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PMC4501523_01 | Female | 50 | A 50 year old woman was admitted to the hospital for bronchoscopic evaluation. She was an active smoker with a cigarette smoking history of 30 pack years and had suffered from repeated bronchitis, shortage of breath and hoarseness as well as repeated episodes of hemoptysis. She also complained of nasal congestion and recurrent sinusitis. A pulmonary infiltrate in the right upper lobe had not resolved after antibiotic treatment.
On clinical examination, stridor was audible in the central airways. The physical examination of other organ systems was not remarkable. Laboratory investigations included normal blood count, normal erythrocyte sedimentation rate and normal biochemistry. A computer tomography scan revealed an atelectasis of the right upper lobe and diffuse nodular thickening of the submucosal tracheal and bronchial walls (Fig. 1). The cartilage showed calcifications. There were no other parenchymal abnormalities. Arterial blood gases, while breathing room air, were normal. Pulmonary function tests revealed normal FVC, FEV1, FEV1/FVC; but decreased PEF and an expiratory plateau in the flow volume loop, suggestive of intrathoracic stenosis (Fig. 2).
Flexible bronchoscopy was performed and a completely irregular surface of the tracheal and bronchial mucosa with prominent white and reddish plaques, extending the lower half of the trachea and main bronchi with reduced airway diameters and diffuse bleeding was found (Fig. 3). Biopsies showed amorphous deposits, identified as amyloid by apple-green birefringence in sections stained with congo red and seen under polarized light. Immunohistochemical staining demonstrated AL amyloid. Systemic amyloidosis was excluded by normal serum protein electrophoresis, normal urine analyses (no Bence Jones protein in the urin), normal rectal biopsy specimens, normal bone marrow aspiration/biopsy, normal echocardiography and abdominal ultrasound.
The patient underwent bronchoscopic debridement with forceps debulking for more extensive bioptic work-up and treatment of airway stenosis. The procedure was complicated by prolonged but self-limited bleeding. The case was discussed at the multidisciplinary respiratory oncology board and it was felt that external beam radiation therapy (EBRT) was an appropriate treatment option. Radiation therapy was explained in detail to the patient because of the scarcity of clinical data regarding its use for TBA. She received EBRT, 20 Gy in 10 fractions. Opposed anterio-posterior and posterior-anterior treatment fields were used to encompass the entire tracheobronchial tree. The treatment volume was determined using CT-guided simulation. Treatment was uneventful. Clinically her symptoms improved and the pulmonary function test stabilized during 12 month follow-up. Chest x-rays at 6 months and CT scans at 12 months after radiation therapy revealed that response to radiation was maintained, with no evidence of disease progression.
Meanwhile the symptoms of chronic sinusitis and nasal congestion worsened. She was transferred to the department of Otolaryngology/Head and Neck Surgery where a CT scan revealed a mass at the bottom of the left maxillary sinus and the nasopharynx. Surgical excision confirmed amyloid deposits submucosally at the roof of the nasopharynx. Functional endoscopic sinus surgery (FEES) was performed to ventilate the left maxillary sinus. To date no further treatment has been necessary. | amyloidosis of the nose, radiation therapy, tracheobronchial amyloidosis | Not supported with pagination yet | null |
PMC269989_01 | Male | 65 | A 65-year-old normally built gentleman presented to the outpatient with sudden onset discharge of faecal contents from an opening in the left inguinal region. The oral intake was adequate and he was passing stools and urine normally. There was no history suggestive of diabetes mellitus, tuberculosis or Crohn's disease. It started as a red, painful swelling in the inguinal region three days back, which ruptured spontaneously discharging faecal contents. Patient had undergone total penectomy with bilateral orchiectomy and perineal urethrostomy, 27 years earlier for carcinoma of the penis, followed by 60 Gy adjuvant external beam radiotherapy (EBRT) to the pelvis and para aortic nodes in 30 fractions. He was on a regular follow-up except for the last five years when he was lost to follow-up.
Examination revealed a well preserved; 140 lbs man (height 165 cm) with no features of anemia, hypoproteinemia, dehydration or electrolyte disturbances. His performance status was good and nutrition risk index (NRI) was >90 (NRI = 1.519 x Serum albumin (g/l) + 0.417 x (current wt/usual wt) x 100). Based on subjective global assessment (SGA) (including clinical criteria like weight loss in previous six months, recent dietary intake compared to the usual intake, presence of anorexia etc.) and Eastern Cooperative Oncology Group (ECOG) criteria his performance and nutritional status was good. Abdomen was scaphoid with no organomegaly or any lymphadenopathy.
There was a discharging fistulous opening in the left ilio-inguinal area approximately 5 cm from the pubic symphysis (figure 1). The discharge was faecal with no evidence of pus in it. The routine investigations including the blood biochemistry were normal. Abdominal ultrasound was normal. Contrast enhanced computed tomography (CECT) abdomen (figure 2) revealed an enterocutaneous fistula and there was no evidence of any recurrent or remnant disease. Fistulogram (figure 3) confirmed the communication with the small bowel. Since the patient was well preserved, with good performance and nutritional status, with adequate oral intake, features of intestinal obstruction, evidence of any underlying malignancy and it being a low output fistula (with a daily out put less than 100 ml), he was managed conservatively on expectant treatment.
The general management included replenishment of fluid and electrolyte losses, and nutritional supplements. The local skin care involved using skin protective emollients creams (containing dimethicone 20%, zinc oxide 7.5%, calamine 1.5% and cetrimide 1.125%) and egg white around the fistula site to protect the skin. An ileostomy bag was fitted in order to allow the fistula to close spontaneously and to measure the output in 24 hours accurately. The daily intake and output chart was maintained to accurately assess the loss for replacement. He made a satisfactory recovery with the output gradually reducing to less than 5 ml and was discharged on the tenth day. Patient has made a satisfactory recovery and now has completed three years on follow-up. | null | Not supported with pagination yet | null |
PMC5448040_01 | Male | 13 | A healthy thirteen-year-old male, who is a competitive basketball and baseball player, sustained a closed, completely displaced fracture involving the medial clavicle after falling off an all-terrain vehicle. He was initially seen in the emergency department, placed in a sling, and referred to the orthopedic service for definitive management. The patient is right-hand dominant and does not smoke. He has no prior history of injury to the right clavicle. At the time of his orthopedic consultation, his pain was described as moderate and sharp localized to the medial end of the right clavicle. The patient reported no numbness or tingling in his arm.
Physical examination revealed a healthy-appearing, pleasant male responding appropriately and in no apparent distress. The pertinent findings on examination included no signs of cervical radiculopathy, no pain or winging of the scapula, intact skin, but significant tenting and swelling over the medial right clavicle, and obvious asymmetry of the right clavicle compared to the left clavicle consistent with a displaced fracture. The end of the clavicle lateral to the fracture site was displaced anterior to the medial component. The remainder of the physical examination revealed no additional injury to the upper extremity. Motor function and sensation were intact throughout the right upper extremity. The brachial and radial pulses were normal and symmetric to the left upper extremity.
Initial radiographs of the right clavicle revealed a fracture involving its medial end without disruption of the sternoclavicular joint (Figures 1(a) and 1(b)). Subsequent CT scan revealed a completely displaced fracture of the right medial clavicle. The fracture site was lateral to the medial physis of the clavicle and without disruption of the sternoclavicular joint. The CT scan allowed for a more definitive characterization of the fracture pattern. Given the rarity with which medial clavicle fractures not involving the physis occur in the pediatric population, a CT scan was desired to provide this information and to help with surgical planning.
Nonoperative and operative treatment options were discussed with the patient and his parents. The factors relevant to pursuing operative treatment included complete displacement at the site of the fracture, desire to obtain anatomic alignment of the fracture to promote healing, potential impact of the displaced clavicle fracture on shoulder function relative to daily activities and overhead sports in basketball and baseball, and clinical outcome allowing the patient to return to activity without pain and restricted function. The operative treatment agreed upon was reduction of the fracture and placement of an elastic intramedullary clavicle nail. Informed consent was signed by the parents and the patient.
At the time of the surgery, a trial of closed reduction revealed an unstable fracture. Subsequently, an incision was made centered over the fracture site. Exposure of the fracture site confirmed complete displacement with the lateral end of the clavicle anterior to the medial end. A drill hole using a 2.7 mm drill was made approximately 1 cm lateral to the medial end of the clavicle. The fracture was reduced and then a 1.5 mm elastic intramedullary titanium nail was passed through the drill hole in the metaphyseal area of the clavicle. The nail was advanced into the intramedullary canal, across the fracture site, and into the lateral end of the clavicle. Placement of the intramedullary nail was confirmed under fluoroscopy (Figures 2(a) and 2(b)). The reduction and fixation were stable as the right upper extremity was brought through a full range of motion. An end cap was placed on the medial end of the clavicle nail to prevent migration. After wound closure and placement of a sterile dressing, the right upper extremity was placed in a shoulder immobilizer. The patient recovered from anesthesia without complications. Preoperative and postoperative examinations of motor function, sensation, and pulses were equivalent.
Postoperatively, the patient was immobilized for 6 weeks until healing was evident on radiographs. He then started active-assisted shoulder stretching exercises at home. Although earlier mobilization could have been considered for this patient, we thought it prudent given his age and nature of the fracture to protect him during the immediate postoperative period for 6 weeks. Given the desire to return the patient to his basketball season and knowing the stress he would put on it during other sports requiring overhead activity of his operative arm, we wanted to ensure complete healing and avoid potential mechanical complications relating to the nail during the early postoperative period. At 8 weeks after surgery, the patient started using his right upper extremity for simple activities of daily living and strengthening exercises. At 10 weeks after surgery, he returned to basketball without any pain or functional limitations. Radiographic images in orthogonal planes confirmed complete healing of the fracture (Figures 3(a) and 3(b)). Approximately 18 weeks after his initial surgery, the patient underwent an uncomplicated procedure to remove the intramedullary nail. Two weeks after intramedullary nail removal, the patient returned to activities without complications. | null | Not supported with pagination yet | null |
PMC9233607_01 | Female | 43 | Our patient is a 43-year-old lady diagnosed with a right parasagittal brain glioblastoma five years ago. The glioblastoma was WHO (World Health Organization) grade 4 with an unmethylated MGMT (methylguanine-DNA methyltransferase) mutation. She underwent gross resection in July 2016 and received concurrent chemoradiotherapy, which was completed in October 2016. In addition, she received adjuvant TMZ (temozolomide) and high-dose atorvastatin as per KFMC ART study protocol, completed in March 2017. Unfortunately, her glioblastoma recurred in February of 2018, for which she underwent reresection and received bevacizumab for five months. Despite these interventions, she developed a third recurrence and required resection in November of 2018, and received bevacizumab and irinotecan for six months. In September 2019, she had a fourth recurrence, which was resected, but she had rapid disease progression and was treated with surgical resection and 35 Gy in 10 fractions of radiation therapy. In December 2019, CT scans showed a fifth recurrence of her disease, which was resected again and treated with reirradiation 35 Gy in 10 fractions until February 2020, when her disease progressed again, and she received bevacizumab and lomustine until August 2020. However, her disease progressed again. She had another surgical resection in September 2020 with reirradiation with 35 Gy in 10 fractions in November 2020. She was maintained on bevacizumab and dose dense temozolomide until May 2021, when she complained of shortness of breath.
The patient was referred to our emergency room for admission. She gave a two-week history of shortness of breath associated with productive cough and right-sided pleuritic chest pain. Before coming to the clinic with her new symptoms, she sought medical advice in a nearby clinic and was given a course of antibiotics with no significant improvement in her condition. Her symptoms progressed to the extent that she had severe dyspnea at rest, could not lie flat, and her cough was worse with blood-tinged sputum. She lost her appetite and had a 6-kilogram unintentional weight loss over two months. She did not have any history of fever or exposure history of note. Subsequently, she presented to our emergency department and was found tachypneic, and her other hemodynamics were normal. The patient looked fatigued, and her physical examination was significant for decreased air entry over the right side on chest auscultation with a dull percussion note in the right lower zone.
Her laboratory investigations showed a WBC (white cell count) of 5.7 x 103/muL and two negative COVID-PCR swabs. Her chest computed tomography showed multiple enlarged necrotic mediastinal and bilateral hilar LN, the largest measuring 4.1 x 2.4 cm. She had a moderate right-sided pleural effusion with multiple bilateral pulmonary nodules, with the largest one in the left lower lobe, measuring 2.4 cm. Multiple lytic lesions in the dorsal vertebrae and a sclerotic focus in the manubrium sternum were identified (Figures 1 and 2).
Bedside thoracentesis was done, draining around 1.2 L of clear yellow fluid initially. The pleural fluid cell count showed 723 x 103/UL WBC with a lymphocytic predominance (78% lymphocytes). Gram stain showed no acid-fast bacilli, and the polymerase chain reaction for mycobacterium DNA was not detected. Pleural fluid cytology showed highly atypical cells, and after sometime, GFAP (glial fibrillary acidic protein) immune staining was positive in some atypical cells. After pleural fluid drainage, the patient's symptoms improved, and she was able to lie flat.
The patient underwent EBUS-TBNA the following day as this case was time-sensitive and to confirm wether she has LN metastasis (which is very rare as mentioned) vs. another primary malignancy or other cause. Five and six needle passes were performed from the right lower paratracheal and right interlobar LNs, respectively. The rapid onsite examination from both stations was consistent with adequate lymph node tissue aquisition and highly suspicious for malignancy, and the samples were sent for further pathological examination and cultures. Cytological smears and cellblock demonstrate clusters of hyperchromatic neoplastic cells with small nucleoli and oval to elongated nuclei in a necrotic background. Cytoplasmic extensions were appreciated in some areas (Figure 3). The tumor cells were negative for broad-spectrum epithelial markers (a panel of keratins was employed). All site-specific epithelial markers were negative. Tumor cells, however, were immunoreactive for GFAP, synaptophysin, and S100 protein (Figure 4). Due to the patient's prior history and immunohistochemical results, the findings were considered consistent with metastatis from the patient's known glioblastoma.
A trial of carboplatin/etoposide was entertained, and the patient received 2 cycles before her extraneural disease progressed. Next-generation sequencing (NGS) was performed on the tissue samples, but no actionable mutations were found. Her performance status declined, and she was transferred under the care of the palliative team in July 2021. Unfortunately, she passed away after several months. Treatment timeline is shown in Figure 5. | null | Not supported with pagination yet | null |
PMC8422441_03 | Male | 68 | A 68-year-old Brazil-nut collector suffered from TBI due to a falling ourico over the right parietal region. He presented at the local emergency department with confusion, headache, dizziness, dysarthria, paresis, and blurred vision. The referral to our hospital occurred only after 3 days, with a GCS of 15. Head CT showed a frontal-occipital subarachnoid hemorrhage and right temporoparietal intracerebral hemorrhage [Figure 3]. The treatment was conservative. | amazon, brazil nut tree, epidemiology, traumatic brain injury, unintentional injury | Not supported with pagination yet | null |
PMC7984903_01 | Male | 74 | A 74-year-old Ecuadorian man who smoked 3 cigarettes a day for a year but quit 15 years prior presented to the Emergency Department (ED) with four days of a productive cough that did not improve with azithromycin. His medical history included hypertension and cataracts. He worked at a clothing factory for years. The body mass index was 24.9 kg/m2. Physical examination showed a blood pressure of 154/86 mmHg, pulse of 115 beats/minute, respiration rate of 18/min, oxygen saturation of 95% on ambient air, and temperature of 101 F. Pulmonary auscultation on hospital admission revealed decreased bilateral breath sounds in the lower lung fields. A chest radiograph showed a large rounded opacity in the superior segment right lower lobe and bibasilar patchy infiltrate. The results of laboratory studies revealed a white blood cell count of 16,000/mul with 82.7% neutrophils and 8% lymphocytes, hemoglobin of 11.9 g/dl, and platelet count of 250,000/mul. Blood and urine cultures yielded no growth, and his acute symptoms of fever and cough subsided with ceftriaxone plus azithromycin. He was discharged home three days after admission. However, his productive cough recurred. More than a month later, computed tomography (CT) scan of the chest revealed multiple, approximately 16.4-28.6 mm, enlarged nodes in the mediastinum and hila, diffuse septal thickening, and multiple ill-defined patchy ground glass opacities with a lower lobe predominance (Figure 1). On transthoracic echocardiography, the systolic pressure of the pulmonary artery was 33 mmHg with normal ejection fraction. The rheumatoid factor was negative, and the antinuclear antibody was positive at 1 : 80 with a homogeneous pattern. The patient was readmitted (2 months after the first episode) due to pleuritic chest pain and dyspnea on exertion. Radiography of the chest revealed bilateral prominent lung interstitial markings particularly in the left perihilar areas, and he was empirically covered for hospital-acquired pneumonia with vancomycin and piperacillin/tazobactam. More than a month later, his C-reactive protein (CRP) was <5 mg/l. He was lost to follow-up for five months but returned complaining of xerophthalmia, and artificial tears provided relief. He was again lost to follow-up for another five months and then noted to have weight loss of 25 pounds over 6 months. Follow-up chest CT findings more than a year after his initial admission included worsening septal thickening and ground glass opacities. Transbronchial lung biopsy demonstrated mild thickening of interstitial septae and increased infiltration by lymphocytes, macrophages, and plasma cells (Figure 2) consistent with LIP. No malignant cells were seen.
Then, he developed bilateral metacarpophalangeal swelling and pain and swelling. To treat LIP, he was started on oral prednisone at a dose of 20 mg/day (0.25 mg/kg). His joint symptoms improved markedly. A laboratory workup was positive for the SSA antibody, and he was diagnosed with Sjogren's syndrome. His CRP was elevated (191 mg/l) and complement levels were low (C3 and C4, 40 mg/dl and 1 mg/dl, respectively). He tested negative for anti-SSB, anti-Jo-1, anti Scl-70, anti-neutrophil cytoplasmic, and anti-double-stranded DNA antibodies. Pulmonary function testing did not show obstructive dysfunction. His postbronchodilator forced expiratory volume in one second (FEV1) was 3 liters (126% predicted), total lung capacity (TLC) was 5.48 liters (104% predicted), and diffusion capacity for carbon monoxide (DLCO) was 17.8 ml/min/mmHg (82% predicted). The patient complained of dry mouth, which improved with pilocarpine. He was lost to follow-up for four months, during which time he discontinued prednisone after five months. Then, he developed anterior uveitis which was treated successfully with topical prednisolone acetate. A follow-up chest CT scan more than a year after oral prednisone was first started showed bibasilar and lingular interstitial fibrotic changes, decreased prominence of associated interlobular septal thickening and ground glass opacity, and a decrease in the size of the lymph nodes. Prednisone 20 mg/day was resumed and slowly tapered over three months. A month after prednisone was tapered to 10 mg, he presented to the ED with oral mucosal bleeding. His vital signs were unremarkable. Physical examination was significant for a bloody oral cavity, bruises on the right arm and legs, and scattered petechiae on the chest and lower legs. Laboratory tests were remarkable for a platelet count of 2000/mul. Peripheral smear did not show schistocytes. CT head was negative for acute hemorrhage. Further labs, including liver function, lactate dehydrogenase, and vitamin B12 levels, were normal. Hepatitis C antibody, hepatitis B surface antigen, and human immunodeficiency virus were negative. No obvious drug-related causes of thrombocytopenia were identified. Blood and urine cultures were negative. A diagnosis of ITP secondary to Sjogren's syndrome was made. A bone marrow biopsy was not done.
The patient was started on prednisone at 80 mg/day (1 mg/kg/d) and received a total of 12 units of platelet transfusion. He was given intravenous immunoglobulin (IVIG) at 1 g/kg/d for two days. His platelet count improved to 84,000/mul by the sixth day, and oral bleeding resolved. The patient was discharged home, but he was unable to continue the prednisone because the prescription was accidentally not transmitted to the pharmacy. After five days, he was readmitted with oral mucosal bleeding. The platelet count was found to be 3000/mul. He received IVIG at 1 g/kg/d and IV methylprednisolone 62.5 mg twice a day. By the fifth day, his platelet count was 178,000/mul. He was discharged on prednisone 80 mg/day, and it was uneventfully tapered off over four months. He was also started on hydroxychloroquine for musculoskeletal pain. His latest platelet count around six months after the last admission was 254,000 cells/mul. | null | Not supported with pagination yet | null |
PMC10423447_01 | Male | 56 | A 56-year-old Caucasian man presented to clinic with a 6-week history of progressive dysesthesia in bilateral arms and legs up to the buttocks and gait difficulties.
He was found to have a multi-focal enhancing and longitudinally extensive lesion with dorsal subpial enhancement on MRI of the cervical and thoracic spine extending from C2 to mid T4 level (Figure 1). Brain MRI was unremarkable. Biopsy of a pulmonary mass showed a granuloma that was not definitive for sarcoidosis. CSF analysis twice revealed persistent pleocytosis (18-61 cells/mm3) and elevated protein but no hypoglycorrhachia. CSF lymphoma markers and ACE levels were unremarkable. Neuromyelitis optica and myelin oligodendrocyte glycoprotein antibodies were also negative. Given the lack of tissue diagnosis and an inconclusive prior lung biopsy, a spinal cord biopsy was done and showed noncaseating granulomas consistent with definite neurosarcoidosis. He was discharged on an oral taper of dexamethasone starting at 15 mg daily along with combination of methotrexate 15 mg weekly and infliximab 5 mg/kg.
Two months later while on this combination regimen of dexamethasone taper, methotrexate 15 mg weekly and infliximab (received 2 infusions), he developed dyspnea and was found to have new pulmonary nodules and cavitary lesions. Simultaneously, he developed right eye blindness. Repeat brain and orbital MRI showed innumerable ring-enhancing lesions in the brain, right endophthalmitis, episcleritis and orbital cellulitis extending from adjacent frontal lesions with surrounding edema concerning for infection (Figure 2). Previous sarcoidosis-related spinal cord abnormalities had resolved completely and immunosuppressants were discontinued. Fungitell test came back positive with a value greater than 500 pg/mL (reference range: <80 pg/mL) and liposomal amphotericin B was initiated. A biopsy of his largest cavitary lung nodule and the vitreous culture of the right eye confirmed Aspergillus fumigatus. Specifically, tuberculosis and malignancy were absent. With a diagnosis of disseminated aspergillosis, amphotericin was transitioned to voriconazole for continued maintenance therapy of disseminated aspergillosis. Unfortunately, he lost all vision in the right eye and underwent enucleation, which substantiated the diagnosis of aspergillosis on histological analysis (Figure 3).
Further surveillance MRIs demonstrated regression of the newly developed lesions, yet persistent contrast enhancement in the brain, despite two years of voriconazole treatment with clinical improvement. Voriconazole was ultimately discontinued due to exfoliative dermatitis, and given his clinical status, no other antifungals were initiated. The patient remains in clinical and radiological remission without immunosuppressive therapy as of 30 months later.
Written informed consent was acquired from the patient for clinical information and medical images to be published. | case report, central nervous system aspergillosis, neurosarcoidosis, tumor necrosis factor-α inhibitors, voriconazole | Not supported with pagination yet | null |
PMC9629952_01 | Female | 20 | A 20-year-old Hispanic woman, living in Laredo, a city on the Mexican border, presented to the emergency room with complaints of recurrent fever, chills, abdominal distension, bilateral lower extremity swelling, nausea, nonbloody emesis, and watery diarrhea. She was discharged from the hospital one week prior, after undergoing a primary low transverse cesarean section at 37.6 weeks of pregnancy. The delivery was performed without any surgical or medical complications. The patient denied any prior medical conditions, use of alcohol or illicit drugs, exposure to known TB contacts, recent travel to Mexico, and a family history of cancer. There are no sick contacts or animals at home.
On initial presentation, the patient was febrile with a temperature of 102.0 F, had tachycardia with a heart rate of 129 beats per minute, and had a blood pressure of 104/56 mm Hg. Physical examination demonstrated abdominal distension and tenderness, shifting dullness to percussion, a positive fluid wave sign, 3+ bilateral peripheral pitting edema, generalized lymphadenopathy and rales and rhonchi in both lungs. The liver and spleen were not palpable. Jaundice, palmar erythema, and spider angiomatosis were not noted. The laboratory reported a microcytic, hypochromic anemia (Hgb = 9.5 g/dL, Ht = 30.8%) with slight anisopoikilocytosis. The platelet count was 305 x 103/mcL and WBC was 7.92 x 103/mcL with 80% neutrophils and 9.3% lymphocytes. Procalcitonin was elevated at 10.68 ng/ml (normal range 0-0.5 ng/ml). CRP was elevated at 13.39 mg/dl (normal range 0.2-0.9 mg/dl) and ESR was elevated at 29 mm/hr (normal range 0-20 mm/hr). Plasma total protein and albumin were reduced at 4.5 g/dL and 2.33 g/dL, respectively. A 24-hour urine protein collection yielded 0.4 g/24 h. Renal function, liver function, and electrolytes were within normal limits.
The presenting X-ray demonstrated diffuse alveolar and interstitial opacities in both lungs, with more extensive findings in the upper lobes and right perihilar region, consistent with pneumonia (Figure 1). The CT of the chest showed extensive bilateral infiltrates with a tree-in-bud configuration (Figure 2). A CT scan of the abdomen/pelvis demonstrated ascites, colonic wall thickening, omental thickening, peritoneal thickening, and mesenteric lymphadenopathy, initially read by radiology as suggestive of carcinomatosis and consistent with the elevated CA-125 level (Figures 3 and 4). There was no hepatosplenomegaly or evidence of cirrhosis. A transthoracic echocardiogram demonstrated a left ventricular ejection fraction of 65% with mild mitral and tricuspid regurgitation. There was no pericardial effusion or evidence of left ventricular thrombus.
The patient was admitted with a presumed diagnosis of sepsis secondary to bilateral pneumonia and ascites of undetermined etiology. The patient was started on treatment empirically with broad spectrum intravenous antibiotics, vancomycin, and piperacillin-tazobactam. Because of persistent tachycardia and fever for several days, vancomycin therapy was switched to doxycycline. Blood tests for syphilis, Rickettsia typhi, Rocky Mountain spotted fever, Q fever, CMV, EBV, HIV, and hepatitis were negative. Blood, urine, and vaginal cultures showed no bacterial growth. Autoimmune antibodies and TB QuantiFERON PCR (QIAGEN, LABCORP) were negative.
Because of the presence of mesenteric adenopathy and concern about a malignant process, tests for tumor markers revealed a mildly elevated CA-125 at 199.5 units/mL (normal range 0-35 units/mL) and an elevated alpha fetoprotein (AFP) at 13.67 ng/mL (normal range 0-5.63 ng/mL). Carcinoembryonic Antigen (CEA), CA 15-3, and CA 19-9 were within normal limits at 0.5 ng/mL (normal range 0-4), 27.4 units/mL (normal range <30) and 23.1 units/mL (normal range 0-37 unit/mL), respectively.
The patient underwent a diagnostic paracentesis which showed a lymphocytic predominance. The calculated serum ascitic albumin gradient (SAAG) was greater than 1.1 and the lactate dehydrogenase (LDH) level in the ascitic fluid was elevated at 399 IU/l. Ascitic fluid culture was negative for bacteria, and cytology was negative for malignant cells.
Subsequently, the Mycobacteria PCR of the ascitic fluid was reported to be positive, and the level of adenosine deaminase (ADA) in the ascitic fluid was reported elevated at 34.9 U/l (normal <7.6 U/l). Around this time, the culture for acid-fast bacilli (AFB) in the sputum was reported to be positive.
Because of the high suspicion of peritoneal TB in this patient, antituberculosis therapy (Rifampin, Isoniazid, Pyrazinamide, Ethambutol (RIPE)) was initiated, prior to the return of the AFB cultures. To confirm the diagnosis of peritoneal TB, the patient underwent a diagnostic laparoscopy with a peritoneal biopsy, which demonstrated an inflammatory response with caseating granulomas and a positive AFB stain, confirming the diagnosis of peritoneal tuberculosis.
After 3 weeks of RIPE therapy, the patient's clinical condition improved and CA-125 decreased from 199.5 units/mL to 89.6 units/mL, CRP decreased from 13.39 mg/dL to 2.55 mg/dl and ESR fell from 29 mm/hr to 23 mm/hr. | null | Not supported with pagination yet | null |
PMC4778176_01 | Male | 19 | A 19-year-old male presented with chief complaints of protrusive lower front teeth and a large lower jaw. Extraoral examination reveals a concave profile, increased lower anterior face height, mild hypoplastic maxilla, positive lip step, hyperdivergent skeletal pattern, and a large mandible [Figure 1a-d]. Intraorally, he exhibits a Class III molar and canine relation, mild crowding of upper and lower anterior teeth, dental midlines matching, and reverse overjet of 3 mm [Figure 2a-e]. A cone beam computed tomography scan (CBCT) reveals the absence of skeletal asymmetry and no abnormality of the temporomandibular joint [Figure 3a-c].
From the clinical presentation and CBCT scan evaluation, it was apparent that the Class III deformity was primarily due to a prognathic mandible in both vertical and anteroposterior planes. A surgery-first approach was planned to perform a bilateral sagittal split setback osteotomy (BSSO) combined with a vertical reduction genioplasty to correct the vertical mandibular excess. In addition, the mildly hypoplastic maxilla was addressed by an autogenic bone transplant.
All teeth were bonded with 0.022" preadjusted brackets (Smartclip , 3M Unitek, St Paul, MN, USA) [Figure 4a-e] and ligated with 0.010 stainless steel ligature wire. A face-bow transfer recorded the relationship of maxilla to the transverse horizontal hinge axis of the mandible [Figure 5]. The patient was subjected to a BSSO set-back and vertical reduction genioplasty of the mandible and autogenic bone transplant of the maxilla. Postsurgically, extraoral images [Figure 6a-d] revealed fulfillment of the surgery objectives. One-week postsurgery, 0.014" NiTi wires were placed in the upper and lower arches [Figure 7a-c], and subsequently, wires were changed to 0.016 x 0.022 NiTi archwires, and the case was finished in 0.017 x 0.025" stainless steel with second-order bends. The overall treatment time was 6 months from start to finish. Treatment results: Six months after surgery, the fixed appliances were removed, and posttreatment photographic images [Figures 8a-d and 9a-e] and CBCT [Figures 10a-c and 11] showed excellent esthetic and occlusal results [Figure 10d]. Pre- and post-treatment superimposition of lateral cephalogram show correction of Class III to Class I skeletal relation. | case series, salient features, surgery-first orthognathic surgery | Not supported with pagination yet | null |
PMC4778176_02 | Male | 21 | A 21-year-old male reported to the orthodontic clinic with chief complaint of protrusive lower front teeth and large lower jaw. On examination, CBCT scan and extraoral images revealed a severe Class III profile with increased lower anterior face height, shallow mentolabial sulcus, positive lip step, and maxillary hypoplasia with no temporomandibular joint abnormality [Figure 12a-g].
Intraoral images revealed a full cusp Class III molar with Class III canine, midline diastema, upper incisor proclination, moderate upper and lower crowding, and reverse overjet of 6 mm [Figure 13a-e].
Bimaxillary surgery with surgery-first approach was planned, wherein a BSSO mandibular setback and Lefort I maxillary advancement would be performed to address the Class III skeletal discrepancy.
All teeth were bonded with 0.022" preadjusted brackets (Gemini , 3M Unitek, St Paul, MN, USA). 0.014Psi NiTi wire were placed in the upper and lower arches [Figure 14a-e] before a BSSO and Lefort I osteotomy of the mandible and maxilla, respectively, and upper bilateral second premolars were extracted. One-month postsurgically, 0.014" NiTi wire was placed. Bilateral upper first premolars were extracted to correct the proclination of upper anterior teeth. Subsequently, the initial wires were replaced by 0.016 x 0.022" NiTi upper and lower arches [Figure 15a-e]. Extraoral photographs at 2 months postsurgery showed good improvement [Figure 16a-d]. Subsequently, the case was finished with 0.017 x 0.025 TMA archwires having second-order bends. The overall treatment time took 9 months from start to finish. Treatment results: Posttreatment photographic images [Figures 17a-d and 18a-e] and CBCT showed great improvements in the profile. Class III facial pattern was changed to Class I profile with pleasing esthetics and good occlusion [Figure 19a-e]. | case series, salient features, surgery-first orthognathic surgery | Not supported with pagination yet | null |
PMC7774431_01 | Female | 17 | Upon returning from a weeklong vacation in New York with her family, 17-year-old Lydia wants to see her friends. "We decide to go to a bar. We drink, we dance, we mess around," shares Lydia (L-14). The night comes to an end, and Lydia and two of her friends accept a ride from a young man they don't know. "He decided to drive at 120 km an hour on a boulevard, after drinking several beers and taking drugs," says Lydia (L-28). Then, while taking a turn, he ran into a tree. "I hit my head. And all the consequences of the traumatic brain injury were horrible," she recalls (L-31). Lydia is brought to a pediatric care center specializing in traumatology, where she wakes up a month later with a diagnosis of severe TBI. "I didn't understand what had happened," states Lydia (L-35). After several months in acute care and intensive functional rehabilitation, Lydia confides: "I still think about it every day" (L-42).
Data collection and analysis yielded six themes and four subthemes, which were considered the most important factors illustrating this family's resilience process: (a) family characteristics (i.e., fighter personality, cultural and spiritual beliefs, presence of hope, keeping a sense of humor), (b) support of family members, (c) support of friends, (d) practicing sports and leisure activities, (e) back-to-school support, and (f) feeling helpful to the adolescent.
I have a strong capacity for resilience (...). When I'm faced with something very difficult, I try to find the positive and adjust my thinking accordingly, then I tell myself: this is what is in front of us, we will try to find a solution with what we have. (D-205)
We got to here, then tomorrow is another day. Then after that, next week, next month, it will be something else. You have to move forward, then you have to move on. Put it behind you, it's over, let's move forward. (D-738)
What helped me, was seeing her [Lydia] every day. Something was always changing (...). I didn't have any expectations, every day, I would see what happens. I lived only 24 hours at a time, I was living day to day and I would embrace every little success. It was a winning strategy. We were all happy. (D-215)
The combative personalities of the family members were one of the subthemes addressed within the family characteristics theme. The strength a family shows when faced with a difficult, or even critical, situation manifests in different ways. For example, Lydia's mother shares:She expands on this capacity she has to not apprehend the future, which, according to her, is a facilitating factor:She adds:For her part, Lydia shared these thoughts, demonstrating her strength of character: "I'm going to finish it [rehabilitation] as quickly as possible (...). Right now I can't walk, but tomorrow, I will!" (L-347). She confides: "I'm happy that my pride took over because that's what helped me persevere" (L-501). "I've been given a second life. A second chance. It's incredible," she concludes (L-694).
Cultural and spiritual beliefs were mentioned. Some of these seemed to be facilitating factors, in the sense that they helped the family adapt to this new situation. Diane states: "I prayed, often, even very often" (D-912).
This family shares, completely naturally and often, the important role that hope played in their resilience process. To wit, Diane states: "We always had hope" (D-630).
A last subtheme identified was keeping a sense of humor despite the situation. Indeed, this seems to have positively impacted this family's journey. On this subject, in reference to Lydia, Diane says: "(...) her sense of humor, she kept her sense of humor, she found humor" (D-593). For her part, Lydia shares: "I kept my sense of humor and this helped a lot, the fact that I always laughed" (L-602). "My accident, I laughed about it because it was easier. It was easier to get past it, and then to joke about it. Even if it made people very uncomfortable, I thought it was funny," she adds (L-999). She also says: "I read somewhere once that "humor shows man's superiority over what he is living ..." (...) this helped me because people stopped treating me like a victim, stopped treating me like I was disabled and needed help" (L-1008).
The family interviewed unanimously stated the importance of the support provided by family members during this difficult situation. Diane says: "I think the fact that family members were there for us, I think that is what made all the difference" (D-279). "My parents were there for me (...), I wouldn't be how I am today without them, without their help, I think that's what made the difference," says Lydia (L-62).
I have a very good friend who is a doctor, so we would talk every day (...). He helped calm me down. This was an incredible source of support because it gave us the energy to keep going. (D-237)
The family expressed the importance of immediate family during this period but also highlighted the positive impact of friends' support. Diane shared her experience:Lydia states that the difficulties would have been even more pronounced "(...) without, also, the help of my family and friends" (L-63).
Taking part in sports and leisure activities can be beneficial not only for the adolescent but also for the parent. In this regard, Diane states: "We have a cottage in the country, this was great therapy for us. It's amazing how much it helped" (D-510). Lydia, when asked, mentioned the benefits of physical activity for her: "I started working out, which really helped me. I feel a lot better. It's when I'm active that I really feel alive again" (L-123). The benefits of her workouts have an impact on her everyday life. In fact, she shares: "When I look at myself today (...), I am the happiest person on Earth. I am so happy" (L-856).
For this family, back to school seems to be an important step because school is an integral part of Lydia's life. Generally, the actions taken by school staff and administration and the support they provide are elements that can facilitate a family's resilience process. In this specific case, Diane assures that the school's support was essential when her teenage daughter went back. "She just wanted to go back to school (...). The school gave her a lighter, adapted course load when she returned" (D-532).
Finally, this family underlined the importance of feeling helpful to Lydia. In this regard, Diane shares: "I would tell the nurses: don't do this, I'll do it (...). My role [is to] provide support: physical and psychological support and understanding" (D-686). | adolescence, case study, humanistic perspective, qualitative design, resilience, traumatic brain injury | Not supported with pagination yet | null |
PMC9900111_01 | Male | 51 | A 51-year-old man with well-controlled HIV (CD4+ count 791 cells/muL and undetectable viral load) on tenofovir alafenamide, emtricitabine, and dolutegravir presented to the emergency room with several weeks of headache, right-sided hearing loss, and memory impairment. He smoked one pack of cigarette per day and marijuana but did not use other drugs. He had lived in the San Francisco Bay Area for over 20 years and was an avid gardener. He did not have a history of incarceration, homelessness, or international travel (Figure 1A).
In the emergency department, he had a witnessed seizure and an MRI of the brain showed swelling of the right cerebral cortex, with abnormal diffusion, T2/FLAIR hyperintensity, and patchy enhancement, and without abnormal susceptibility weighted imaging (Figure 1B). The imaging was consistent with meningoencephalitis, and serial lumbar punctures (LP) revealed a neutrophil-predominant pleocytosis (WBC: 260-756/muL with 12-70% neutrophils), mild hypoglycorrhachia (nadir 33 mg/dL), and elevated protein (76-95 mg/dL). Gram stain and bacterial culture in CSF were negative, as was cryptococcal antigen. He was started on empiric valacyclovir and broad-spectrum antibiotics.
After 3 weeks of inpatient antimicrobial treatment, the patient failed to improve. Comprehensive serological and cerebrospinal fluid (CSF) testing for infectious etiologies was non-diagnostic including serum toxoplasmosis IgG and CSF fungal cultures, microbial antigen and antibody testing, VDRL, universal bacterial, fungal, and mycobacterial PCR, and metagenomic next-generation sequencing (mNGS). Diagnostic biopsy of the leptomeninges overlying the right cerebrum revealed dense connective tissue with a mixed inflammatory infiltrate including granulocytes, and B and T cells (Figure 1C), suggestive of an infectious etiology. The underlying cortical brain tissue was also biopsied and showed no significant pathological abnormality. Histochemical stains for acid-fast bacillus (AFB), Grocott's methenamine silver stain (GMS), and period acid-Schiff (PAS) stains on the brain tissue were negative, arguing against mycobacterial or fungal infection and Whipple's disease. A Quantiferon Gold TB test was negative. However, given the insensitivity of CNS TB diagnostics, the lack of clinical improvement, and a persistent neutrophil-predominant CSF pleocytosis with mild hypoglycorrhachia, he was started on empiric therapy for TB meningoencephalitis with isoniazid, rifampin, ethambutol, and pyrazinamide and transferred to an outpatient rehabilitation facility.
One week after discharge (week 7), the patient presented to the emergency department with new-onset expressive aphasia, hyponatremia, and ataxia. EEG revealed right greater than left diffuse background slowing and disorganization without epileptiform activity. Repeat LP again showed a neutrophilic pleocytosis (WBC 194/muL with 50% neutrophils), protein 75 mg/dL, elevated IgG index (0.7, ref. <=0.6), and five restricted oligoclonal bands (OCBs). Brain MRI showed persistent meningoencephalitis with new ventriculomegaly. A large volume LP was performed. The opening pressure was 25 cm and the patient regained his speech after 1 h later, had improved gait, and improved mental status. He proceeded to get a ventriculoperitoneal shunt and continued on anti-TB therapy.
A CSF autoimmune encephalopathy panel was reported as positive for NMDA receptor autoantibodies (1:8 tissue titer) as well as putatively non-specific additional staining on tissue. The whole body PET and scrotal ultrasound were negative. A repeat autoimmune encephalopathy panel was NMDA receptor antibody positive on cell-based assay but negative on reflex tissue staining. Because the clinical phenotype was markedly inconsistent with anti-NMDAR encephalitis, empiric anti-TB therapy was initially continued. Nonetheless, an autoimmune etiology was increasingly considered as a result of the repeatedly negative infectious workup and lack of improvement in antibiotic therapy. In this context, he received a 5-day course of methylprednisolone 1 gram daily on week 8 followed by a prednisone taper with dramatic improvement in his memory and language. Anti-TB therapy was ultimately discontinued after ~1 month of treatment.
Over the following months, attempts to taper off prednisone resulted in clinical and radiological deterioration. Surveillance brain MRIs demonstrated worsening of meningoencephalitis with a new focus on enhancement in the left amygdala. A second brain biopsy of glucocorticoids at month 10 showed severe granulomatous and suppurative meningoencephalitis with a perivascular and intraparenchymal lymphocytic infiltrate, diffuse astrogliosis, and microglial activation. The pattern of inflammation was again suspicious for mycobacterial infection; however, AFB and AFB-FITE stains and AFB culture of the brain tissue were negative, as were mycobacterial universal PCR and metagenomic next-generation sequencing.
Three weeks after the second biopsy, the patient was retreated with IV methylprednisolone followed by oral prednisone maintenance with subsequent clinical improvement over the next few months. Repeat imaging 3 months after the resumption of glucocorticoid treatment demonstrated near the total resolution of leptomeningeal enhancement and non-progression of his encephalitis. The patient was then transitioned empirically to mycophenolate mofetil and successfully weaned off prednisone without recurrence of his meningoencephalitis. He remained seizure-free on antiseizure therapy. Repeat CSF at 26 months post-presentation showed normal cellularity (3 WBC/muL with 87% lymphocytes and 17% monocytes), normal glucose (57 mg/dL), mildly elevated protein (67 mg/dL), persistent elevated IgG index (0.7 ref. <=0.6), and two oligoclonal bands. MRI brain every 6 months showed stable multifocal T2/FLAIR cortical and white matter hyperintensities without associated enhancement. Following 3 years of immunosuppressive therapy, 4.5 years after his initial presentation, mycophenolate mofetil was stopped and the patient remained stable. Neurological assessment documented fixed sequelae consisting of dysarthria, executive dysfunction, and episodic memory impairment. | ank3, ankyring, autoantibody, autoimmune, axon initial segment (ais), human immunodeficiency virus (hiv), meningoencephalitis, node of ranvier | Not supported with pagination yet | null |
PMC7298523_01 | Male | 88 | An 88 year-old gentleman with good performance status, presented to clinic with chronic cough. A computed tomography (CT) scan of his thorax revealed a 5.3 x 4.7 x 5.7 cm left lower lobe mass with abrupt cut off at the posterior-basal segment of the left lower lobe (Fig. 1). He underwent bronchoscopy with radial endobronchial ultrasound (EBUS)- guided TBLB via a transoral route under moderate sedation with intravenous midazolam and fentanyl. A concentric heterogenous lesion with hyperechoic dots was identified on radial EBUS in the posterior-basal segment of the left lower lobe (Fig. 2). No visible endobronchial lesions or stenosis were seen. Forcep biopsies were performed under fluoroscopy to obtain 8 tissue samples. Post-procedure chest radiograph did not reveal any pneumothorax and he was discharged home. Histology of his lung mass confirmed lung adenocarcinoma with epidermal growth factor receptor (EGFR) exon-19 mutation detected.
He was admitted 6 days after his procedure, with symptoms of fever and cough that started since his bronchoscopy. Chest radiograph showed a new left lower lobe lung collapse with left pleural effusion. He was commenced on empirical intravenous antibiotics. CT scan of his thorax, abdomen and pelvis revealed an interval development of a moderate left pleural effusion with abscess formation within the lung mass (Fig. 1). In addition, a liver lesion consistent with metastatic disease was seen. Bedside ultrasound confirmed a left loculated pleural effusion, which was drained. Pleural studies showed an exudative picture based on Light's criteria with no cell count predominance, and cytology did not reveal any malignant cells. Pleural fluid cultures grew Streptococcus anginosus. Peripheral blood cultures were negative for any bacterial growth. A dental review showed no signs of abscess or dental infections. Our patient subsequently opted for prolonged antibiotic therapy and pleural drainage, and declined intrapleural fibrinolysis or surgical decortication in view of his advanced age. He made a full recovery following a 6 week course of Augmentin. Erlotinib was commenced prior to discharge for stage 4 lung adenocarcinoma. | empyema, lung abscess, transbronchial lung biopsy | Coronal CT images of the left lower lobe lung mass pre-bronchoscopy , and. |
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PMC5905859_01 | Male | 78 | A 78-year-old man with no significant history was found to have prostate cancer stage cT2cN0M0, Gleason score of 4 + 5 with a serum prostate-specific antigen value of 8.05 ng/cc in June 2016. He underwent RALP with extended lymph node dissection. The operation time was 339 minutes and bleeding volume was 150 cc. The Foley catheters were removed on postoperative day 6, according to the usual protocol. No urinary leakage from the anastomosis sites was observed. He was discharged without event on postoperative day 14. Pathologic diagnosis was pT3b, Gleason score of 4 + 5, and positive resection margin. Lymph node metastasis was not detected.
On postoperative day 25, he presented with fever and general fatigue. Physical examination showed body temperature of 38.2 C, regular pulse (84/min), blood pressure 145/84 mm Hg, and a distended abdomen. Initial laboratory findings were a normal white blood cell (WBC) count and increased C-reactive protein level (11.2 mg/dL). Liver and renal functions were normal. Computed tomography (CT) showed massive ascites and extensive thickened peritoneum (Fig. 1). CT of lungs showed slightly worsening granular shadows and a mucous plug in the left upper lobe (Fig. 2). Initial ascites sample was cloudy, with increased WBCs and normal creatinine level, precluding urinary leakage. Bacterial infection of a lymphocele was considered and cefmetazole 2 g/day for 3 days was prescribed but was ineffective. Blood, urine, sputum, and ascites cultures were negative.
Repeat abdominocentesis revealed 2900 WBCs/mm3 (87% lymphocytes) and adenosine deaminase of 140 IU/L (normal 0-33 IU/L). Ziehl-Neelsen staining and initial polymerase chain reaction (PCR) for M. tuberculosis were negative. Interferon-gamma release assay was indeterminate. Abdominocentesis was performed repeatedly, attempting to diagnose peritoneal TB. The fourth ascites sample was positive. The cultures and PCR of urine, sputum, and gastric lavage for TB were negative. The patient was found to have peritoneal TB. Anti-TB therapy with isoniazid (INH) (300 mg/day), rifampicin (RFP) (450 mg/day), ethambutol (EB) (1000 mg/day), and pyrazinamide (PZA) (1200 mg/day) was initiated. His temperature dropped immediately, and his general condition improved (Fig. 3). After the initial 2-month phase, INH and RFP were given for another 4 months. Three months after oral treatment completion, there was no recurrence of infection or ascites. | complication, extended lymph node dissection, peritoneal tuberculosis, robot-assisted laparoscopic prostatectomy | CT of the left upper lung:. preoperative, and. |
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PMC4980689_02 | Unknown | 12 | An open trial showed the efficacy of CoPs treatment in adults with tics compared to waitlist with a 6:1 ratio. Results showed that 10 of the 85 participants completely reduced tic onset after therapy (gains maintained at 6-month follow-up). Prior results also showed efficacy in tic reduction in adults with or without medication, following CoPs treatment. The therapy was applied to five adolescents with TD, in a pilot study. Results showed a decrease in tic frequency and intensity and improvement in social functioning for the five participants. The CoPs treatment has also been adapted for children with TD addressing explosive outbursts (EO). Results showed a decrease of EO frequency of at least 34% for four participants out of six. Another participant showed a 75% decrease in posttreatment, but did not complete the follow-up assessment. The last participant showed a 67% decrease between the beginning of therapy and follow-up, despite an increase of EO frequency at baseline assessment. Finally, a single-case design study of the CoPs treatment addressing tic severity in childhood was conducted with 11 children aged 8-12 years old. A decrease of 29.8% of tic onset was observed posttreatment (p < 0.001, d = 0.97), and the decrease was monitored over 1 year. Results showed a decrease of at least 1 SD in measures, post 12 months.
After this pilot study, a manualized version of the treatment protocol in children termed Facotik has been finalized, and the aim of the current study is to evaluate its efficacy in a larger consecutive case series. Based on previous research, a decrease of tic severity was expected after treatment. The efficacy of the treatment adapted for children will have important implications for the intervention in TD and whether addressing the underlying sensorimotor processes is sufficient to reduce tics. | tourette disorder, children, cognitive–behavioral therapy, psychophysiological, tics, treatment | Not supported with pagination yet | null |
PMC3573823_01 | Male | 75 | A 75-year-old Japanese male was admitted to our hospital complaining of lumbago, which had persisted for one month. He was an ex-smoker, and had type II diabetes, hypertension and ileus. Chest computed tomography (CT) revealed a 37-mm diameter mass lesion in the left lower lobe of his lung. The histological diagnosis of the transbronchial biopsy specimen was adenocarcinoma (fig. 1a). We diagnosed the patient to have lung cancer with multiple bone metastases, brain metastasis, and liver metastasis (cT2aN0M1b, stage IV). After the patient provided informed consent, we analyzed the specimen obtained from the TBLB for EGFR mutations. The analysis identified an 18-base pair deletion in exon 19 (fig. 1b). The patient was treated with cisplatin and pemetrexed as a first-line chemotherapy; however, after one cycle of the treatment, he developed paralysis of his left leg, and this symptom was rapidly progressive. Therefore, an emergency operation was performed for posterior lumbar interbody fusion. As second-line chemotherapy, the patient was started on 150 mg/day erlotinib. One month later, the size of both the primary lesion and the bone metastases were found to have decreased.
Three months later, he developed dyspnea at rest and edema. Furthermore, he had severe dyspnea and orthopnea, with a resting heart rate of 160-170 beats/min and a blood pressure of 63/43 mm Hg. His temperature was 36.8 C, and his arterial oxygen saturation was 88.0% on room air. A physical examination revealed distant heart sounds and a significantly distended internal jugular vein to the jaw level. An electrocardiogram revealed sinus tachycardia and borderline low-voltage QRS complexes. Chest radiographs showed cardiomegaly with an increased cardiothoracic ratio and bilateral pleural effusion (fig. 2a). A CT scan of the chest revealed a large pericardial effusion (fig. 2b). An emergency echocardiogram confirmed the presence of a large pericardial effusion, together with evidence of diastolic collapse of the right artrium, ventricle and left atrium, consistent with cardiac tamponade (fig. 2c).
The patient underwent pericardiocentesis of 520 ml of malignant sanguineous fluid, and placement of a pericardial drain, while having removed 1,100 ml of sanguineous fluid, in which adenocarcinoma was detected (fig. 3a). EGFR mutation analysis was performed in this pericardial effusion, and both the 18-base pair deletion in exon 19 and a second mutation, T790M in exon 20, were detected (fig. 3b and c). The patient temporarily recovered from the state of shock and survived for one month after drainage. He could not undergo further chemotherapy because his general condition had worsened. He died due to progressive disease five months after the original diagnosis. No autopsy was performed. | cardiac tamponade, epidermal growth factor receptor mutation, non-small cell lung cancer, pericardial effusion, t790m | Not supported with pagination yet | null |
PMC3964670_01 | Female | 48 | In April 2011, after positron emission tomography/computed tomography (CT/PET) to investigate a residual solid right lower lobe lung nodule following pneumonia, a 48-year-old woman was referred to a thyroid surgeon with an incidental thyroid lesion (Fig 1). She had a two-month history of throat discomfort but no voice dysfunction or difficulty breathing. Significant past medical history included right thyroid lobe surgery for a degenerative colloid adenoma 19 years previously. She had never had any thyroid hormone dysfunction and there was no family history of thyroid disease.
On examination, she was clinically euthyroid. There was a visible 3cm nodule in the lower pole of the left lobe of the thyroid. There was no associated lymphadenopathy. Thyroid function tests were normal. Ultrasonography demonstrated a 3.75cm circumscribed, solid, echogenic nodule in the lower pole of the left lobe of the thyroid and radiological appearances suggested a follicular lesion. There was regrowth of the right thyroid remnant. Fine needle aspiration showed follicular cells that were graded as Thy3 (intermediate risk of malignancy).
After discussion with the patient about the alternative management options and in light of the undiagnosed lung lesion, she underwent an urgent left hemithyroidectomy and excision of the right thyroid remnant. There were no post-operative complications.
Pathology of the excised nodule revealed the index lesion was a benign thyroid adenoma. There was an incidental microcarcinoma measuring 0.8mm in the specimen. This was completely encapsulated and not adjacent to the thyroid capsule. The presenting lung lesion was ultimately determined to be unrelated to the thyroid lesion. Resection of the lung nodule revealed an area of necrotising granulomatous inflammation. There were no acid-fast bacilli or fungal bodies seen and there was no clear clinical indication of tuberculosis. | null | Not supported with pagination yet | null |
PMC2640408_01 | Male | 40 | A 40-year-old man, diagnosed with Addison's disease 2 years before, presented with a 1-month history of intermittent fever and no other relevant symptoms. His physical examination revealed no abnormalities. A haemogram, a routine biochemistry profile and coagulant tests were all normal, except for an erythrocyte sedimentation rate of 70 mm/hour. Radiographs of the chest and abdomen and an echocardiogram were unhelpful. Brucella spp serology and blood cultures were negative. Mantoux (5 UI) was 12 mm. Ziehl-Neelsen stain and mycobacterial urine cultures were negative, as was a bone-marrow culture for Mycobacteria and Brucella. An abdominal computer tomography (CT) scan showed a hypodense mass in the right lobe of the liver and an enlarged right adrenal gland with calcifications. Fine-needle aspiration of the liver was negative for malignant cells. Microscopic examination of stained specimens revealed no acid-fast bacilli or other microorganisms, and cultures (aerobic and anaerobic) for bacteria and fungi were negative. Laparotomy was performed, obtaining purulent material from the adrenal gland, with extension to the liver and duodenum. Gram and Ziehl-Neelsen stains of the pus were negative, but Streptococcus constellatus was isolated in pure culture, using the API 20S system. The minimum inhibitory concentration (MIC) for penicillin was 0.03 microgr/ml. Microscopic examination revealed granulomas in the liver and adrenal tissue. The patient was treated with ceftriaxone, rifampicin, isoniazid and pyrazinamide, and his condition improved. Lowenstein culture of the abscess material yielded positive result for Mycobacterium tuberculosis. | null | Not supported with pagination yet | null |
PMC2640408_02 | Female | 68 | A 68-year-old woman was admitted to hospital with general malaise, arthromyalgia and fever lasting for 2 months. Ten days prior to admission, she started experiencing dorsolumbar pain radiating to the right rib region. The only abnormalities observed on physical examination were temperature of 37.5 C and pain on palpation of the spinal thoracic apophyses 11 and 12. The results of her blood analysis were as follows: haemoglobin 13.4 gr/dl, 7100 leucocytes/mm3, erythrocyte sedimentation rate 39 mm/hour and albumin 3.3 gr/dl. Other blood tests results were normal. Mantoux was 25 mm (5 UI). Brucella serology and blood cultures were both negative. An echocardiogram showed hypertensive cardiomyopathy. Radiography of the spinal column revealed destruction and crushing of thoracic vertebrae 10 and 12, with an adjacent paravertebral mass. Spinal CT scan showed destructive and erosive lesions in those vertebrae, involving the intervetebral discs and an adjacent paravertebral mass.
CT-guided fine-needle aspiration of the paravertebral mass was performed. Purulent material was extracted, for which both Gram and Ziehl-Neelsen stains were negative. Subsequently, Streptococcus constellatus was isolated in pure culture, using the API 20S system. MIC for penicillin was 0,06 microgr/ml. The patient was treated with penicillin with initial clinical improvement. After 2 months of treatment, her condition suddenly worsened both clinically and radiologically. On physical examination, two soft lumps were detected in the left paravertebral region near the level of thoracic vertebra 11 and pus was extracted. Gram stain revealed no microorganisms and Ziehl-Neelsen stain showed acid-fast bacilli. Concurrently, the culture from the needle aspiration performed during the first admission was received, with an isolation of Mycobacterium tuberculosis. Treatment was initiated with rifampicin, isoniazid and pyrazinamide, resulting in clinical improvement. One month later, the patient was admitted to the hospital with acute myocardial infarction and died a few hours later. | null | Not supported with pagination yet | null |
PMC2640408_03 | Female | 70 | A 70-year-old woman presented with a constitutional syndrome lasting 2 months with pain in the lumbar region and a limp. Physical examination revealed temperature of 38 C and pain on flexing the left hip but no other abnormal findings. Blood analysis showed: haemoglobin 11 gr/dl, 13800 leucocytes/mm3 (75% neutrophils), 380000 platelets/mm3; erythrocyte sedimentation rate 109 mm/hour, fibrinogen 981 mg/dl, albumin 2.9 gr/dl and ferritin 559 ng/ml. Radiography of the abdomen showed blurring of the line of the left psoas. Abdominal CT scan revealed a mass of lower density in the left psoas.
Percutaneous drainage was placed in the above-mentioned area, and purulent material was extracted. Gram-positive cocci in chains were observed, while a Ziehl-Neelsen stain was negative. Subsequently, Streptococcus anginosus was isolated in pure culture, using the API 20S system. MIC to penicillin was 0.03 microgr/ml. Treatment with ceftriaxone was initiated and the drainage maintained for 1 week.
When a new CT scan conducted after two weeks of treatment showed persistence of the abscesses, surgical drainage was indicated. Forty-five days after admission, Lowenstein culture from the previous puncture yielded a positive result for Mycobacterium tuberculosis. Treatment with isoniazid, rifampicin and pyrazinamide was initiated, after which the patient's condition improved both clinically and radiologically. | null | Not supported with pagination yet | null |
PMC6942756_01 | Male | 58 | A 58-year-old male underwent elective left lower lobe resection via video-assisted thoracoscopic surgery (VATS) for a 9 mm lung nodule suspicious for malignancy. The patient's past medical history included hypertension, gastroesophageal reflux disease, depression, anxiety, chronic hepatitis C, and AUD. Liver biopsy and Fibroscan had not been performed to assess for cirrhosis, preoperatively. His last admission was six months prior to surgery for alcohol withdrawal syndrome. The patient reported drinking 10 beers a day with his last drink on the day before surgery. He denied tobacco, but reported using marijuana. He had no known allergies or adverse drug reactions.
His baseline liver function tests (LFTs) before surgery were: AST 111 U/L, ALT 50 U/L, total bilirubin (TBili) 0.5 mg/dl, INR 1.11 (Table 1). AST/ALT ratio suggested alcoholic hepatitis. Nutritional status was marginal, with albumin 3.1 g/dL. Platelet count was 120,000 k/cmm. Outpatient medications included aripiprazole, sertraline, and a multivitamin.
There were no hypotensive episodes or other complications during VATS. Blood loss was less than 50 ml. He received general anesthesia with sevofluorane. There were no other concomitant medications given intraoperatively or postoperatively that could have contributed to deteriorating hepatic function.
The following analgesics were given to optimize pain control postoperatively: APAP 1 g orally every six hours for three days, oxycodone 5-10 mg orally every four hours as needed, and hydromorphone 0.5 mg IV every four hours as needed for breakthrough pain.
On postoperative day 2 (POD 2), the patient remained hemodynamically stable. On POD 3, he developed nausea and discomfort in the right upper quadrant. LFTs were checked and noted to be elevated: AST 3169 U/L, ALT 758 U/L, TBili 2.8 mg/dL (Table 1, Figure 1). He had received a total of 9 g of APAP over a period of 3 days, with no more than 4 g over a 24-hour period. APAP was discontinued at this time due to LFT elevation and concern for iatrogenic hepatotoxicity.
On POD 4, LFTs peaked: AST 5417 U/L, ALT 1372 U/L, TBili 3.5 mg/dL; albumin 2.6 g/dL, INR 2.83, lactate 6.1 mmol/L; and glucose 58 mg/dl. Renal function and arterial blood gas remained within normal limits. Urine toxicology screen was negative. The patient reported right-sided abdominal pain and nausea, and had two episodes of emesis. He was noted to have scleral icterus and grade I encephalopathy. He had a syncopal episode secondary to hypoglycemia, which promptly responded to intravenous dextrose administration. A CT scan of the abdomen and pelvis was done, revealing fatty liver without portal vein thrombosis or other biliary tract abnormalities. Ischemic hepatitis (shock liver) was thought unlikely as the patient was never hypotensive.
The hepatology service was consulted and a diagnosis of acetaminophen-induced hepatotoxicity was suspected based on the clinical picture, laboratory values, and negative imaging. However, APAP level was within normal limits at 12.9 mg/L (normal therapeutic range 5-20 mg/L). Despite a nontoxic APAP level, NAC was recommended. On POD 4, he was treated with 3 weight-based doses of IV NAC: 9 g (150 mg/kg) over one hour, then 3 g (50 mg/kg) over four hours, followed by 6 g (100 mg/kg) over sixteen hours. A one-time dose of oral vitamin K 10 mg was also administered.
On POD 5, APAP level decreased to 0.5 mg/L and LFTs were noted to be declining. He was discharged on POD 7, at which time he was clinically stable and asymptomatic. LFTs continued to improve and the patient's renal function remained at baseline and within normal limits. One month after discharge, LFTs further decreased to: AST 427 U/L, ALT 82 U/L, TBili 2 mg/dL.
Throughout the patient's hospital stay, renal function remained at baseline (0.8-1.4 mg/dl). | null | Not supported with pagination yet | null |
PMC9826551_01 | Male | 42 | A 42-year-old male presented to our outpatient clinic with a 2-year history of the right ankle stiffness, swelling, and intermittent catching and locking sensation. The patient's medical history and family history were unremarkable. There was no history of trauma. Before the onset of symptoms, he played football regularly.
On physical examination, there was a marked swelling of the right ankle. Passive and active dorsal and plantar flexion were reduced compared to the left ankle. Pain was elicited at extreme passive dorsiflexion and with direct palpation over the anterior ankle. The hyperplantarflexion test produced pain over the posterior aspect of the ankle and feeling of locking. The laxity observed with the anterior drawer maneuver was similar to that of the left ankle. Subtalar motion and midfoot motion were unrestricted and non-tender. Neurological and vascular examinations were normal.
Typical clinical findings on physical examination suggested anterior and posterior ankle impingement syndromes as most likely causes of patient's symptoms, indicating further radiological investigations. Standard anteroposterior (AP) and lateral (LL) radiographs (Fig. 1) showed diffuse swelling of the ankle and osteophytes of the anterior distal tibia and dorsal talar neck. Os trigonum and unexpected soft-tissue mass were detected in the posterior ankle recess, behind the posterior process of the talus. Magnetic resonance imaging (MRI) of the ankle was performed to evaluate the soft-tissue mass (Fig. 2). MRI demonstrated a nodular lobulated soft-tissue mass in the posterior ankle recess measuring 29 x 18 x 19 mm. T1- and T2-weighted images showed heterogeneous mass, with predominantly low-to-intermediate signal intensity. PD-weighted images revealed a high signal intensity halo surrounding the low intensity signal mass. Furthermore, an additional bone fragment, which was not visible on standard AP and LL radiographs was detected on MRI. It was impinged between distal portion of tibia and fibula. Multislice computed tomography of the ankle was also performed to define the morphology of the bony lesions and help more accurate planning of surgical procedure.
The patient's history, clinical, and radiological findings helped to confirm the diagnosis of anterior and posterior ankle impingement syndromes with the presence of a loose body, impinged between tibia and fibula. Because of typical MRI findings for LPVNS, an excisional biopsy of the soft-tissue mass in the posterior ankle recess was indicated to confirm the diagnosis. Due to concurrent presence of anterior and posterior ankle pathology, we decided to use combined two-portal endoscopic hindfoot approach and anterior ankle arthroscopy within the same operative session. The procedure was executed with the patient under spinal anesthesia and in the prone position. A tourniquet was placed around his upper leg but was not inflated. This was done due to good experience of senior authors, who have previously described that not inflating the tourniquet, while using an arthroscopic pump, provides good visualization and good postoperative results in anterior ankle arthroscopy. The hindfoot endoscopy was performed through the posteromedial and posterolateral portals as described by van Dijk et al., and by utilising a 4.0 mm 30 arthroscope. The portals were used interchangeably as viewing or working portals. At the level of subtalar joint, a well-encapsulated, pedunculated, and yellowish-brown nodule was observed. It was cut from the stalk with a radiofrequency wand and the mass was removed from the joint with a grasper (Fig. 3). The specimen was sent for histopathological analysis. Partial synovectomy was then performed at the base of the lesion with the help of a shaver and a radiofrequency wand to achieve complete macroscopic local clearance. Os trigonum and a loose body impinged in the interval between tibia and fibula were also extracted from the posterior ankle recess. After the completion of hindfoot endoscopy, the portals were sutured. The patient was then turned into a supine position, leaving all instruments on a sterile table. The foot and ankle were disinfected again, with new sterile draping applied. Once the patient was adjusted in the supine position, anterior ankle arthroscopy was performed using standard anteromedial and anterolateral portals utilizing a 2-portal dorsiflexion method. An arthroscopic shaver and a radiofrequency wand were used to debride hypertrophic synovium and fibrotic tissue in the anterior ankle recess, including the lateral and medial gutters. A combination of a shaver and a burr was then used to reshape the anterior distal tibia and dorsal talus to their native form. At the end of the procedure, a No. 12 closed suction drain was placed through the anterolateral portal into the joint and the portals were sutured.
The drain was removed on the 1st postoperative day, when patient started with active and passive range of motion (ROM) exercises. A posterior splint with the ankle in the neutral position was used overnight for 3 weeks after the surgery. Weight-bearing as tolerated, with the support of crutches, was allowed for the first 3 weeks after surgery. It was followed by gradual progression to full weight-bearing over the next 3 weeks. During this period, patient underwent a course of physiotherapy, consisting of a ROM and strengthening exercises.
Histopathological analysis confirmed that nodular mass found in the posterior recess of the ankle was indeed a LPVNS (Fig. 4). No recurrence was noted at the final follow-up, 2 years after the surgery. Patient was satisfied with the procedure. He continued playing football several times a week, without any restrictions. | synovitis, ankle, arthroscopy, hindfoot endoscopy, pigmented villonodular | Not supported with pagination yet | null |
PMC2259312_01 | Female | 65 | A 65-year-old multi-gravida presented with complaints of abdominal pain and postmenopausal bleeding per vagina for seven months. Her past medical history was insignificant with no history of tuberculosis, inflammatory conditions of the uterus or iatrogenically introduced substances in uterus. She had attained menopause about 15 years back.
Gynecological examination revealed atrophic ectocervix flushed with vagina. The vagina appeared normal. Uterus was bulky of around 12 weeks and adnexae were unremarkable. Colposcopy showed schiller's unstained areas on anterior lip of cervix. Ultrasound abdomen showed endometrial thickness of 2.6 cms. and fluid in the endometrial cavity. Ectocervical biopsy showed strips of moderate to severe dysplastic stratified squamous epithelium. Endometrial curetting revealed strips of stratified squamous epithelium showing moderate dysplastic changes. No normal endometrium was seen. The pyometra was drained, following which the patient underwent type-II radical hysterectomy. The procedure was well tolerated and the postoperative period was uneventful. | null | Not supported with pagination yet | null |
PMC8039304_01 | Male | 79 | A 79-year-old man was presented to our hospital with 1-month cough and fever. He has visited two other local hospitals for more than three times where a series of examinations were performed. His chest computerized tomography (CT) scan revealed ground-glass nodules in the upper lobe of the right lung with multiple enlarged lymph nodes in the mediastinum which were suspected of tumors (Figure 1A). In order to clarify the nature of the lesion he further took a needle biopsy of lymph nodes under bronchoscope while pathological results showed no evidence of tumors. Presumed to be pulmonary infection, he was then given piperacillin-tazobactam, moxifloxacin, and empirical anti-tuberculosis treatment successively. His body temperature gradually returned to normal, but the cough still exists. One week later, his cough worsened without fever. Outpatient blood routine examination revealed that white cells count (WBC) 13.1 x 109/L, C-reactive protein (CRP) 183.51 mg/L. Re-examination of chest CT showed infectious lesions in both lungs, left hilar shadow increased, and mediastinal lymph nodes slightly enlarged (Figure 1B). For further treatment, he was admitted to the Department of Infectious Diseases of Ningbo First hospital. His initial diagnosis was bacterial pneumonia.
He has not taken any immunosuppressants or glucocorticoids before. He had hypertension for more than 10 years treated by amlodipine and bisoprolol, diabetes mellitus for 3 years treated by insulin with unsatisfactory glycemic control, and eczema for 10 years without therapy. He did not have chronic airway disease although he had smoked for 20 years and quitted smoking for more than 20 years. Three years ago, he had undergone Permanent Cardiac Pacemaker implantation. As a Ningbo native, he had retired from a Metal Instrument Factory where he worked as an installer of metallic parts for more than 20 years. And he hasn't been to the wild recently. He has no prior family history of malignancy, infectious diseases or psychosis. Physical examination showed large eczema-like scars on two legs and found no other positive signs.
After admission, we examined his cellular immune function for lymphocyte subsets analysis (Table 1), immunoglobulins, cytokines, and found no obvious immune deficiency. And his tuberculosis T-SPOT. TB test, human immunodeficiency virus (HIV) test, tumor biomarkers (including carcinoembryonic antigen, carbohydrate associated antigen 199, alpha-fetoprotein, and cancer antigen 125), Antinuclear antibody (ANA) and Antineutrophil autoantibodies (ANCA), serum cryptococcal antigen colloidal gold test, and serological test for syphilis were all negative.
Since he has used broad-spectrum antibiotics with poor efficacy, we performed meropenem, tigecycline consecutively (Table 2). However, the peak temperature did not drop (Figure 2), and the inflammation indexes did not improve (Table 3). During hospitalization, his galactomannan (GM) was positive in serum. Thus, we empirically add caspofungin for antifungal treatment. Meanwhile, we replaced tigecycline with linezolid. Since then, the fever seems to have gradually eased but WBC increased slowly (Figure 3). We ordered another chest CT scan which indicated the lesions of pneumonia were larger than before and pleural fluid appeared (Figure 1C). On November 5, he underwent a fiberoptic bronchoscopic examination (Figure 4) and lavage of the basal segment of the left lower lobe. Bronchoalveolar Lavage fluid (BALF) was tested for mNGS, culture, liquid-based cytology. At the same time, blood sample was sent for mNGS examination.
Four days later, we changed antibiotics to intravenous voriconazole and piperacillin-tazobactam since mNGS suggested Penicillium marneffei infection (Table 4). Moreover, we prolonged sputum culture time to one week and the fungus was further validated by culture (Figure 3). After he was prescribed targeted anti-fungal agent of voriconazole 200 mg twice daily, the clinical presentations and laboratory indicators were gradually returning to normal (Table 3). The whole process was concluded (Figure 5).
Prior to follow-up, he carried on taking voriconazole 200 mg twice daily regularly without relapse. On December 9th, his repeated chest CT scan showed that pneumonia was cured and pleural effusion were absorbed obviously (Figure 1D). | anti-fungal treatment, case report, immunocompetent, next-generation sequencing, talaromyces marneffei | Not supported with pagination yet | null |
PMC10315836_01 | Female | 0 | Table 1 displays a comparison of scores on the EMQ between T.I., C.I., and the overall sample mean. T.I.'s gross motor score was similar to both C.I.'s score and the sample mean at the 3-month visit but substantially lower by the 24-month visit. Most notably, T.I.'s 3 month fine motor score (0) was more than 3 standard deviations below the sample mean (M = 0.67, SD = 0.20). A score of 0 indicates that the infant has not yet demonstrated any of the fine motor behaviors listed on the questionnaire. Examples of fine motor behaviors that other 3-month-old infants demonstrated were: "opens the fingers of each hand spontaneously," "brings hands together near the face, chest, or tummy," and "tightly holds onto a toy placed into his/her hand". T.I.'s mother verbally noted at the 2.5-month visit that T.I. liked to tuck his thumb into the palm of his hand. The experimenter noted on a study documentation form that she had trouble opening T.I.'s fingers to place a rattle in his hand.
Results from video coding (quantitative analyses) revealed that T.I. spent less time manually engaging with presented objects than C.I. at the first three visits (see Figure 2).
At the 8.5-month visit, T.I. engaged in more rhythmic play than C.I. (e.g., repeatedly sliding the activity ball across the table; see Figure 3C). T.I.'s mother noted during the filmed session that T.I. liked to slide his hands and toys across the table at home. In contrast, the second author noted that C.I. spent more time manipulating the individual components of the activity ball (see Figure 3D).
At 2.5 and 3 months, T.I. demonstrated a longer latency to visually attend to presented objects compared to C.I., as measured by the number of seconds that passed between the presentation of an object and the onset of the first "look" code. When the experimenter attempted to direct T.I.'s attention to an object, T.I. often took up to 10 s to visually orient. C.I. did not demonstrate this same behavior and tended to orient quickly to new objects (Figure 3B). The second author noted that T.I. displayed a strong preference for looking down at his hands and at the table's surface (Figure 3A). Compared to C.I., T.I. rarely shifted his attention-looking at his hands, the toy, and the table surface continuously for up to 60 s. At the 8.5-month visit, T.I. no longer looked at his hands, but similarly maintained visual attention to presented objects for periods of up to 60 s. In contrast, C.I. frequently shifted his attention between the toy, his mother, and the experimenter.
At the 8.5-month visit, the experimenter stacked a set of 5 blocks to build a tower and asked the infant to imitate this behavior. Neither infant successfully stacked any blocks. As with the activity ball, T.I. slid the blocks back and forth across the table. The same blocks task was repeated at the 24-month visit. Both C.I. and T.I. successfully built a block tower. C.I. proceeded to knock down the tower, just as the experimenter had demonstrated. In contrast, T.I. accomplished the same goal by removing each block from the tower one at a time to create a straight line of blocks on the table.
A second imitation task at the 24-month visit involved inserting a metal spoon into the side of a lightbox to activate a display of lights. C.I. inserted the spoon into a hole in the side of the lightbox, just as the experimenter demonstrated (see Figure 3F). In contrast, T.I. explored the box itself and discovered that the top of the box could be removed to reveal the lights inside (see Figure 3E). No other child in the study noticed the removable top, which was designed to be discreet (i.e., all painted the same color). T.I. then used his finger, instead of the tool, to activate the light, a solution that was not modeled by the experimenter. | autism, emulation, fine motor skills, infancy, visual attention | Not supported with pagination yet | null |
PMC5107655_01 | Female | 37 | A female 37-year-old patient was admitted to our Department in January 1991. A left complete and a right partial staghorn stone had been diagnosed 8 years before. She had not wanted therapy. In May and June 1990, she had had pneumonia and she had lost 10 kg weight in the last 6 months. She was cachectic with a body mass index (BMI) of 15.8 (159 cm, 40 kg). She had no flank pain, but 39.6 C fever. Urine showed all signs of urinary tract infection (UTI) and the urine culture was positive for Escherichia coli with resistance against 8 of 16 tested antibiotics. The white blood count was 11.400 and she was anemic with an Hb of 8.9 g/dL. Her creatinine was 1.5 mg% (0.6-1.3 mg%). Actual plain film and CT showed bilateral complete staghorn stones. There was no visible dye excretion on the left side in the external intravenous urography, and sonography showed hydronephrosis of the left kidney with nearly no parenchyma. In the split isotope function test from June 1990, the left kidney had only 26% function and in January 1991, it was only 13%.
Her temperature came gradually down to below 37 C during 11 days of appropriate antibiosis, the Hb was improved with transfusions, and the creatinine was 1.2 mg%.
On Monday, January 21, 1991, after 2 days without fever, right PCNL was done by an experienced associate: puncture of the lower right calix, dilation to 26F, Amplatz sheath, ultrasound disintegration of the lower caliceal extension and the pelvic portion of the struvite staghorn stone. The middle and upper caliceal extensions were left for secondary extracorporeal shockwave lithotripsy at the end of that week, which was the strategy at that time. There was venous bleeding upon removal of the Amplatz sheath, which stopped after placement of a 22F balloon nephrostomy. The patient was extubated, transferred to the post-acute care unit, and from there during the night to the intensive care unit. The next day, she was reintubated because of respiratory problems. The right kidney functioned well with a urine production of between 100 and 200 mL/hour, while the left kidney produced between 0 and 30 mL/hour. Despite appropriate therapy, her condition worsened and the right kidney, which was thought to be a persistent septic focus, was removed on January 26th. She died on February 1, 1991 due to multiple organ dysfunction.
I do not remember how we discussed the case in our 6-month fatality conference. When you lose a patient, you start thinking about what you could have done different. Your thoughts are orientated toward your past experience and to other urologists' thoughts published in the literature.
What was the cause and what could alternative options have been or be in a similar case?
To be able to predict complication of PCNL, Moreno-Palacios et al. designed an index based on an analysis of 354 PCNLs with four cases of (Clavien 5) deaths and four cases of (Clavien 4) sepsis. A high Charlson comorbidity index (CCI) was most important, predicting a Clavien >=3 grade complication.
Our patient was at least among the moderate risk group, but the risk cannot be calculated as a low BMI of 15.8 does not fit into the CCI. Would it help to know the risk? Would palliative care have been an option?
Benson et al. reported on a special series of 219 patients treated between 2007 and 2012. Nearly, a third of them had lower urinary tract reconstruction or neurogenic bladder, which are known to have a complicated course after PCNL.
Group 1 patients had preprocedural nephrostomy for 19.6 days and antibiosis for an average of 13.7 days, while group 2 had 6.8 days of preoperative antibiosis and access established on the day of surgery.
None of the patients with preprocedural nephrostomy had post-PCNL sepsis and drainage was the only factor preventing it. Two third of the patients with prolonged antibiosis and nearly 3 weeks of nephrostomy drainage still had positive stone cultures. This well-known problem to sterilize the upper urinary tract is difficult if not impossible to manage.
In a similar series with neuromuscular disorders, preliminary drainage between 1 and 62 days, but not prolonged antibiosis, reduced the incidence of bacteremia/sepsis from 5/19 to 0/16.
Reports dealing with aspiration of purulent urine at the time of the puncture saw similar frequencies of septic complications with or without temporary percutaneous drainage and advocated same session PCNL when the patients had no other signs of infection preoperatively.
Many other reports on the benefit of decompression of the upper urinary tract deal with obstruction due to ureteral stones and already manifested systemic inflammatory response syndrome (SIRS)/sepsis, do not add to a better understanding of our case.
Preliminary urinary diversion by uni- or bilateral percutaneous nephrostomy would have been an option. My personal experience is that even the puncture to place a small emergency nephrostomy tube for drainage only, sometimes provokes bouts of fever. The drainage, especially for complete staghorn stones, is rarely complete and there are areas left without drainage, without the possibility to recognize these areas and change the conditions.
We could have tried this approach or maybe we should have tried it.
Zhao et al. looked at the advantage of a staged PCNL procedure. Patients were treated either in planned two sessions (group 1) or in one intended session (group 2) between 2011 and 2013. Patients with UTI were treated with antibiotics for 3-5 days until the culture turned negative. A second stage was done after 3-5 days. There were ~50% complete stags in both groups and 30% had UTI and infection stones. The results favored the planned two sessions, with SIRS or septic shock happening in 11 (7.5%) group 1 and 22 (15.8%) group 2 patients. Of those patients with infection stones, 22% of group 1 and 41% of group 2 had infection-related complications.
Our patient was treated in a planned two-session procedure.
Al-Kohlany et al. were the first to publish a prospective randomized controlled study comparing a temporary series (2001-2003) of 79 patients and 88 complete staghorn stones treated by open surgery (OS) vs PCNL. Unusually, few patients, 15/88 had UTI and only 5 of the 50 stones analyzed were composed of struvite. The intra- and postoperative complication rate of 15/43 for PCNL and 31/45 for OS favored PCNL. Postoperative sepsis was equally distributed with three cases in each group.
Lunardi et al. did 26 anatrophic nephrolithotomies between 2005 and 2013. Half of the patients had preoperative UTI and only two cases of pyelonephritis were among the few complications. A meta-analysis on PCNL and laparoscopic surgery (LS) for pelvic stones showed principal advantages for the latter with a lower incidence of bleeding, less postoperative fever, and a higher stone-free rate, but the analysis was not focused on staghorn stones.
A more recent publication by Aminsharifi et al. compared 45 adults who underwent PCNL, OS, or LS between 2010 and 2015.
Active UTI was an exclusion criterion and all patients had antibiotic treatment 1 day before surgery, "so that sterility of the urine was ensured before the operation." Only 11 patients had struvite stones. For PCNL, they used a 30F Amplatz sheath. Anatrophic nephrolithotomy was used in all other patients.
Only a few patients needed transfusion (three OS, two PCNL, and one LS) and there were no postoperative infectious complications. A significant reduction of renal function in all groups was most pronounced in the open group from 42% to 33%. In the lap-series of Pastore et al., all nine patients had a negative preoperative urine culture and received a single dose of broad-spectrum antibiosis. All nine patients were PCNL failures and none of them had struvite stones. After laparoscopic pyelolithotomy and laser disintegration of the peripheral stones through a flexible scope, there were no complications and only one residual stone of 9 mm.
In 1991, OS could have been an option, but it was and is not stylish; laparoscopy was and probably would not be an option today.
Mortality after PCNL is rare. Unsal et al. saw three mortalities in their multicenter report on 1406 procedures. Desai reported no mortality in their 773 cases of PCNL for staghorn stones and in the Croes report with 5335 cases, of which 1466 (27.5%) had staghorn stones, mortality was not mentioned.
The above patient is the only fatality in our PCNL report with a mortality rate of 0.3% and is the only patient I have ever seen dying after PCNL.
Mortality is rarely presented in more than a few words in the urological literature and not as a major topic, especially not in urolithiasis articles. A rare and laudable exception is the article "Mortality and flexible ureteroscopy: analysis of six cases" by Cindolo et al. However, mortality is not infrequent. A nationwide U.S. sample showed 46,354 patients being hospitalized in 2009 with infected urolithiasis. Nearly 4000 and 1500 had associated sepsis and severe sepsis, respectively. Nine hundred twenty-seven patients had died in 2009 compared to 592 in 1999. The percentage of sepsis or severe sepsis in patients with kidney stones is less than half of those with ureteral stones, but concerning mortality, the percentage is identical. Probably 1000 people die each year in the United States from infected urolithiasis, but may be only a handful make into a publication.
So is it difficult to learn from our fatal complications or is it impossible because death will come anyway?
We cannot learn a special procedure or a technical detail from these rare cases, but may be a strategy. It is the physician who finds himself in an emergency situation of a personal relationship. The question is not how to remove the stone or how to find a solution, which is focused on the urological problem, but to get access to a human being. It is an emergency situation not only for the patient but also for the surgeon, because he will never have a chance to read what will happen. Other qualities than those of an urologists are required.
"The steamer toiled along slowly on the edge of a black and incomprehensible frenzy. The prehistoric man was cursing us, praying to us, welcoming us:who could tell? We were cut off from the comprehension of our surroundings; we glided past like phantoms, wondering and secretly appalled, as sane men would be before an enthusiastic outbreak in a madhouse. We could not understand, because we were too far and could not remember, because we were traveling in the night of first ages, of those ages that are gone, leaving hardly a sign:and no memories.
The earth seemed unearthly. We are accustomed to look upon the shackled form of a conquered monster, but there:there you could look at a thing monstrous and free. It was unearthly, and the men were:No, they were not inhuman. Well, you know, that was the worst of it:this suspicion of their not being inhuman. It would come slowly to one. They howled, and leaped, and spun, and made horrid faces; but what thrilled you was just the thought of their humanity:like yours:the thought of your remote kinship with this wild and passionate uproar. Ugly, yes, it was ugly enough; but if you were man enough you would admit to yourself that here was in you just the faintest trace of a response to the terrible frankness of that noise, a dim suspicion of there being a meaning in it which you:you so remote from the night of first ages:could comprehend. And why not? The mind of man is capable of anything:because everything is in it, all the past as well as all the future."
Heart of darkness is still in the board's and in the reader's list of the 100 best novels. However, it has also been criticized to promote prejudice against Africa. I have read the novella again as a grown up and for me, it is a perfect example for the different views on reality and for the forces of fate:black or white. | heart of darkness, joseph conrad, mortality, percutaneous nephrolithotomy | Not supported with pagination yet | null |
PMC9510984_01 | Male | 49 | A 49-year-old Asian male who was diagnosed with hepatitis B virus (HBV)-positive HCC with no evidence of macrovascular involvement underwent curative segment VII liver resection (3.8 cm x 2.8 cm) 85 days after testing COVID-19 negative using RT-PCR swab test. A second patient, a 45-year-old Asian male diagnosed with invasive stage II T3N0 cecal adenocarcinoma (right-sided CRC), underwent laparoscopic right hemicolectomy 9 days after testing COVID-19 negative using RT-PCR swab test. Patient details have been reported previously. The two patients had mild symptoms of acute respiratory infection and were hospitalized for isolation purposes; in addition, they were unvaccinated and did not require oxygen therapy or any medical treatment for COVID-19. Importantly, the patients had not undergone any cancer treatment before COVID-19 infection or surgery, ruling out immunological effects caused by other therapy. While the CRC patient did not have other co-morbidities, the HCC patient had chronic HBV and past TB infections.
Initially, we conducted spatial transcriptomic (ST) profiling on paired specimens consisting of tumor and adjacent normal tissue from the HCC and CRC patients. This analysis was performed on fresh frozen sections using the Visium Spatial Gene Expression (10x Genomics) assay. The tissue sections placed on the capture areas of the Visium slide were fixed, stained with hematoxylin and eosin, and imaged. After tissue permeabilization, mRNA molecules were reverse transcribed to generate spatially barcoded cDNA molecules, which were then PCR-amplified and sequenced at 50,000 reads per capture spot. All bioinformatic analysis was performed on individual tissues using the sctransform-normalized data computed by R package Seurat (v4.0.3) (unless stated otherwise) ( Supplementary Figure 1A ).
Using the Visium ST data ( Supplementary Figure 2 ), we carried out spatial enrichment analysis (AddModuleScore function in Seurat) based on the SARS-CoV-2 immune response signatures from two public databases ( Supplementary Table 1 ). In the first database, Lee et al. identified lymphocyte-associated (CD8+ T cells and NK cells) and myeloid-associated (monocyte and dendritic cells) COVID-19 signatures, using influenza patients as a reference. In the second, Ren et al. determined three lymphocyte-associated (B cell, T cell, and NK cell) COVID-19 signatures and one myeloid-associated (neutrophil) signature, using COVID-19-negative peripheral blood mononuclear cells (PBMCs) as a reference. For the four lymphocyte signatures, spatial profiles were broadly consistent in all four tissues analyzed ( Figure 1 ); however, some differences in localization were observed for the SARS-CoV-2 response signatures associated with T cells and NK cells in the CRC-adjacent normal tissue ( Figures 1L, P ). By contrast, myeloid-associated SARS-CoV-2 responses did not demonstrate distinctive spatial localization, except in the HCC-adjacent normal tissue ( Supplementary Figure 3 , see panels B and F ).
Independent of these SARS-CoV-2 immune responses, we identified four regions with distinct ST niches within each tissue using a spatially-aware clustering method (BayesSpace R package v1.2.0) ( Figures 2A-D ); the four clusters were selected arbitrarily to balance resolution and interpretability. Generation of uniform manifold approximation and projection (UMAP) plots was based on the top 30 principal components (RunPCA and RunUMAP functions in Seurat) of the ST niches ( Supplementary Figure 4 ). The distribution of SARS-CoV-2 immune responses was consistent in the ST niches in the HCC tissues but heterogenous in the CRC tissues ( Supplementary Figures 5 and 6 ).
As other viral infections (e.g., HBV in the HCC patient) can induce similar immune responses, we further localized SARS-CoV-2 specific immune responses in tissues. This analysis was performed by quantifying SARS-CoV-2 spike protein (SP) counts on serial tissue sections, using RNAscope ( Figures 2E-H ; brightfield images presented in Supplementary Figures 7 - 8 , raw counts given in Supplementary Table 2 ; Supplementary Materials ). The SARS-CoV-2 SP counts were spatially variable, especially in the CRC tumor tissue ( Figures 2I-L ). Despite the variability, the viral-high regions (defined as the ST niches exhibiting the highest average SARS-CoV-2 SP counts) were confirmed by mIHC performed on FFPE sections ( Supplementary Figure 9 ; Supplementary Materials ). Both RNAscope and mIHC analyses were based on multiple high-quality regions of interest (ROIs; marked as boxes in Figures 2E-H and Supplementary Figures 9A-D ) that were selected by a pathologist (JY). To compute ST niche-average counts, the images were manually overlaid onto the Visium tissue images, and the ROIs were assigned to the closest ST niches ( Supplementary Table 2 ; average SP and NP counts of ROIs assigned to individual ST niches are shown in Figures 2I-L and Supplementary Figures 9E-H ). Furthermore, using a pathologist (JY)-trained machine-learning classifier in QuPath (v0.3.2; Supplementary Materials), the viral-high regions were predominantly identified as stromal or normal epithelial tissues ( Supplementary Figure 10 ).
Subsequently, we conducted immune cell type analysis using the microenvironment cell population (MCP)-counter (MCPcounter R package v1.2.0), whereby the presence of a cell type in the Visium spots was defined using a threshold of MCP-counter-estimated log2 expression > 0. The results revealed intra-tissue heterogeneity in terms of tissue coverage (% spots) and detectability ( Figure 2M ). B cells were detected homogeneously within viral-high regions in all tissues (although they were relatively sparse in the HCC-adjacent normal tissue), while other immune cells were either sparse or dispersed throughout the tissue ( Figure 2M , Supplementary Figures 11 - 12 ). To identify the immune cells that elicited the SARS-CoV-2 immunological effect, we examined the association between the presence of immune cells and public COVID-19 signature scores. Analysis of the COVID-19 signatures identified by Lee et al. showed that within viral-high regions, the lymphocyte-associated SARS-CoV-2 response was significantly positively associated with the spatial localization of B cells across HCC and CRC tissues (P < 0.005, Figures 3A-D ); in contrast, the myeloid COVID-19 signature was only significantly associated with T cells in HCC tissues (P < 0.005, Figures 3A, B ). Similar findings were obtained using the COVID-19 signatures from Ren et al.; the lymphocyte-specific SARS-CoV-2 responses (i.e., B, T, and NK-associated responses) were significantly associated with the spatial localization of B cells across HCC and CRC tissues (P < 0.005), but not CRC-adjacent normal tissue ( Supplementary Figure 13 ).
To further dissect the specificity of B cell subsets involved in the tissue-localized lymphocyte-based COVID-19 response, we characterized and annotated individual B cell phenotypes (memory [MBCs], naive, intermediate, and plasmablasts) by single-cell analysis of ex vivo SARS-CoV-2-stimulated patient-matched samples. Stimulated lymph node samples (CRC) had been obtained previously, while PBMCs from the HCC patient were stimulated following the same procedure prior to analysis (Supplementary Materials). Single-cell sequencing used Chromium Single Cell 5' and 3' Reagent Kits v2 and v3 (10x Genomics, San Francisco, CA, USA), while paired-end sequencing (2x150 bp) was performed using the NovaSeq 6000 platform (Illumina, Inc., San Diego, CA, USA) (Supplementary Materials; bioinformatic analysis is depicted in Supplementary Figure 1B ). Marker genes for the B cell phenotypes were identified by comparing SARS-CoV-2-stimulated and unstimulated samples (using the FindMarkers function in Seurat; Supplementary Table 3 ). The above-identified patient- and SARS-CoV-2-specific B cell phenotypes were then mapped to individual Visium spots using robust cell type decomposition (RCTD package v1.2.0), whereby the presence of a cell type was recorded if detected as a singlet or a doublet. Within the viral-high regions, plasmablasts and MBCs exhibited the highest spot coverage in HCC and CRC tissues, respectively ( Supplementary Figures 14 - 15 ); however, only the spatial localization of MBCs demonstrated a consistently positive association with lymphocyte-associated SARS-CoV-2 immunological effects ( Supplementary Figures 16A-H ). Notably, MBC infiltration (measured by spot coverage) was higher in the viral-high regions of CRC tissues, compared with the viral-high regions of HCC tissues ( Supplementary Figure 14 ).
We evaluated the potential impact of a persistent SARS-CoV-2-induced B cell immune response on spontaneous or immunotherapy-induced anti-tumor activity in situ by determining the immuno-predictive scores (IMPRES). IMPRES, a transcriptomic biomarker for ICB, was computed by summing the binary outcomes (gene 1 > gene 2, using spot-level log-normalized counts) of 15 pairs of immune checkpoint genes ( Supplementary Table 4 ) and normalizing by the available pairs. In individual tissues, the spatial localization of B cells exhibited variable associations (positive and negative) with IMPRES scores across the ST niches. However, within viral-high regions, spots with detection of B cells showed consistently higher IMPRES scores for all four tissues ( Figure 4 ; viral-high regions are indicated by orange stars/red arrows). By contrast, spots with detection of T cells and cytotoxic lymphocytes (representing key effector cells in immunotherapy) demonstrated consistently higher IMPRES scores across the ST niches, although they were undetected in some CRC regions. Although the differences in scores were not significant, they were indicative of overall trends. CD8+ T cells were generally sparse and not detected in any of the viral-high regions of the four tissues. Other immune cells, including NK cells, myeloid dendritic cells, monocytic lineage cells, and neutrophils, were either not positively associated with IMPRES scores or were undetected in the viral-high regions.
We also evaluated another transcriptomic predictor of ICB, the tumor immune dysfunction and exclusion (TIDE) framework, which simultaneously measures T cell dysfunction and exclusion as key mechanisms of tumor immune evasion. TIDE analysis was conducted on the web platform , along with the analysis of individual immune components. Specifically, T cell-dysfunction/exclusion, interferon gamma (IFNG), Merck18 (T-cell inflammatory), microsatellite instability (MSI), and tumor-associated macrophages (TAMs; M2 subtype) were analyzed based on ST niche-average log-normalized counts (normalized by log-normalized counts of all other ST niches as a reference). While spatially heterogeneous TIDE scores were observed across the ST niches, TIDE-predicted ICB responsiveness (TIDE scores < 0) was detected in the viral-high regions in both HCC and CRC tumor tissues ( Supplementary Figure 17A ). Notably, compared with other ST niches, the viral-high regions in both HCC and CRC tumor tissues harbored the highest T cell dysfunction, IFNG, and Merck18 signatures ( Supplementary Figure 17B ), alongside the lowest T cell exclusion, microsatellite instability (MSI), and TAM M2 signatures ( Supplementary Figure 17C ). In addition, the viral-high region in HCC tumor tissue harbored the highest expression of the checkpoint molecule CD274 (PD-L1) and the lowest immune cytolytic activity; however, such immune exhaustion was not replicated in the viral-high region of CRC tumor tissue, which might be due to the shorter time elapsed since infection ( Supplementary Figure 18 ). Immune cytolytic activity scores were obtained by averaging the sctransform-normalized values of two key cytolytic effectors, granzyme A and perforin. Mapping of HBV transcripts (based on 73 HBV variants downloaded from NCBI GenBank; Supplementary Table 5 ) onto ST niches revealed that HBV was detected homogeneously across the HCC tumor tissue but was relatively sparse within the viral-high region; in addition, HBV was not detected in the adjacent normal HCC tissue ( Supplementary Figure 19 ).
All analyses were conducted in R (version 4.1.0). Associations were evaluated by the non-parametric Kruskal-Wallis test. Statistical significance was judged using a two-sided alpha significance level of 0.05. | sars-cov-2, case report, immunotherapy, intra-tumor heterogeneity, spatial transcriptomics, tissue-localized immunity | Not supported with pagination yet | null |
PMC9568322_01 | Female | 3 | A 3-year-old Hispanic girl with no prior history of systemic disease was sent for a consultation to our uveitis service for the further evaluation of her bilateral granulomatous uveitis. Her family history was remarkable for PsA in her father and uncle. The review of systems revealed that she had a 3-month history of a skin eruption and left knee arthralgia.
Upon a comprehensive examination, her best-corrected visual acuity (BCVA) was 20/60 in both eyes. The intraocular pressure was 13 mmHg in the right eye (OD) and 14 mmHg in the left eye (OS). A slit-lamp exam revealed bilateral band keratopathy, large mutton-fat keratic precipitates (KPs), multiple posterior synechiae (Figures 1(a)-1(d)), and 4+ anterior chamber cells. The fundus view was very poor for both eyes due to the presence of band keratopathy and limited pupillary dilation. A B-scan ultrasound was unremarkable, with no evidence of vitreous opacity.
The physical examination was remarkable for left knee edema and a right axillary nonpruritic eruption consisting of irregularly shaped erythematous papules and plaques with thin scale (Figure 2(a)). No dactylitis, enthesitis, or nail changes were identified. As the differential diagnosis of granulomatous iridocyclitis includes infectious and noninfectious etiologies, a diagnostic work-up was ordered, and the patient was started on hourly topical prednisolone acetate 1% and atropine sulfate 1%, the latter, twice a day.
The radiological images were remarkable only for left knee effusion. The girl's chest X-ray was normal, with no lymphadenopathy, opacities, or cavitations. Routine laboratory tests revealed an erythrocyte sedimentation rate of 80 mm/h and an unremarkable complete metabolic panel (CMP) (Na: 141 mEq/L, K: 3.7 mEq/L, Cl: 103 mEq/L, blood urea nitrogen: 9.00 mg/dL, and serum creatinine: 0.25 mg/dL) and urinalysis (negative for glucose, protein, red blood cells, white blood cells, and casts). Her immunology work-up was remarkable for a positive ANA test (1 : 1,280), with a homogenous pattern and negative anti-double-stranded DNA antibody, anti-Smith antibody, rheumatoid factor (<10 IU/mL), and cyclic citrullinated peptide IgG. Fluorescent treponemal antibody absorption (FTA-ABS), venereal disease research laboratory (VDRL), and tuberculosis interferon-gamma release assay (IGRA) tests were negative. Serological tests for the human leukocyte antigen B27 haplotype, HIV, hepatitis A IgM, hepatitis B core IgG, and the hepatitis C antibody were negative.
A punch biopsy of a skin lesion revealed psoriasiform acanthosis, thin supra-papillary plates, neutrophils in the stratum corneum, and superficial perivascular lymphocytic infiltrate (Figure 2(b)). Based on these results, the uveitis specialist concluded a diagnosis of bilateral granulomatous iridocyclitis associated with early-onset JPsA.
After establishing a tissue diagnosis of JPsA, oral prednisolone (1 mg/kg) was added to her therapy. Following 2 weeks of treatment, her uveitis was inactive, bilaterally. The BCVA improved to 20/40 in OD and remained stable at 20/60 in OS. At the 2-month follow-up visit, her uveitis remained inactive, and her BCVA improved to 20/30 in OD and 20/40 in OS. At this visit, a standardized prednisolone tapering protocol was begun, and she was started on subcutaneous methotrexate (0.2 mg/kg/week) as a corticosteroid-sparing therapy. | null | Not supported with pagination yet | null |
PMC6481261_01 | Female | 57 | A 57-year-old woman was admitted to the trauma center for severe TBI after she jumped out of a window. Initial physical examination revealed a Glasgow coma scale score at 3, and her pupils were dilated and nonreactive. She also presented with hemorrhagic shock with diffuse facial bleeding. She was quickly stabilized after transfusion and infusion of norepinephrine. The whole-body computed tomography (CT) scan showed depressed skull fractures, bilateral subdural hemorrhage, intraventricular hemorrhage, diffuse subarachnoid hemorrhage, right frontal contusion, and diffuse brain swelling (Fig. 1). The CT angiography showed a right anterior cerebral artery rupture with blush next to the frontal contusion (Fig. 2). An aneurysm was not present. There was no other vessel injured. There were also multiples facial fractures. Given the severity of the injuries, neurosurgeons did not perform an emergent surgical decompression. Despite advanced medical care, the patient developed refractory intracranial hypertension leading to brain death the fourth day after the admission.
Blunt cerebrovascular injuries are not uncommon after blunt trauma. Recent studies found an incidence of approximately 1-2%. Severe TBI is a well-known risk factor of BCVI, with an incidence up to 5-fold higher in this specific population. However, catching active bleeding due to an intracranial arterial injury is exceptional. BCVI occurs mainly in large extracranial vessels like carotid and vertebral arteries. Moreover, most of these cases concern low-grade injuries according to Denver scale. The Denver scale (or Biffl scale) is the most commonly used grading scheme in the literature and has been reused in the 2010 guidelines of the Eastern Association for the Surgery of Trauma to guide treatment (Table 1). In severe TBI, high-grade injuries (grades IV and V), as in the present report, represent only approximately 20% of the cases.
Secondly, this case emphasizes the crucial importance of early detection of BCVI in TBI patients because of its therapeutic impact. Indeed, BCVI is potentially devastating because they can lead to neurovascular events. Untreated BCVIs of the carotid an vertebral arteries could have an overall stroke rate approaching 40 to 60%, and a stroke-related mortality rate as high as 50%. Recent studies showed that medical therapy with anticoagulation or antiplatelet agents that is started before the onset of neurological symptoms could reduce the occurrence of neurological events by a factor of 6, decreasing the stroke rate as low as 4% in grade I to III patients. Some practitioners could have a doubt concerning the safety of this therapy in patients with severe TBI. However, studies including individuals with neurological injury (TBI or spinal cord injury) did not show an increased risk of intracranial hemorrhage progression after early initiation of pharmacological treatment. In the case of high-grade injuries (grades IV and V), endovascular interventions with coiling could be of interest.
The modalities of BCVI screening evolved greatly over the past 2 decades. Digital four vessel subtraction angiography has been considered the standard imaging modality. Nowadays, CT angiography using 32-channel and higher multidetector CT scanners has supplanted subtraction angiography as a first-line screening modality, with a sensitivity of 66-98%, a specificity of 92-100% and a good cost-efficiency ratio. MR imaging is currently not considered an appropriate tool for BCVI screening. However, it allows a good vessel wall imaging and may be considered for follow up BCVI evaluation.
To our knowledge, this is the first report of a complete anterior cerebral artery transection with free contrast extravasation. This report emphasizes that screening of BCVI may be essential after a severe TBI if there is a suspicion of vascular injury on the initial noncontrast CT scan, and even more with the widespread availability of high-performance CT scanners which permits a fast, efficient and low-cost examination of vascular structures. | anterior cerebral artery, bcvi | Not supported with pagination yet | null |
PMC8116590_01 | Male | 31 | A 31-year-old Chinese male with sudden onset of blurred vision in his right eye for 2 days was referred to our clinic on January 21, 2019. On examination, best corrected visual acuity (BCVA) was 20/20 and 20/1,000 in the left and right eye, respectively. Furthermore, intraocular pressure (IOP) was elevated to 39.3 mmHg in the right eye. A slit lamp demonstrated ciliary hyperemia, positive fresh keratic precipitates (KP), positive anterior chamber flare and cell, and positive relativistic afferent pupillary disorder (RAPD) in the right eye. On fundus examination, the right eye showed prominent white sheathing of retinal venules and, to a lesser extent, arterioles in all the quadrants, resembling the appearance characterized as "frosted branch angiitis." Supratemporal retinal hemorrhages indicated concomitant branch retinal vein occlusion (BRVO). Moreover, macular edema and disc hyperemia were presented (Figure 1A). Fundus photograph of the left eye showed normal appearance (Figure 1B). Optical coherence tomography (OCT) suggested macular edema (Figure 1D). Fluorescein angiography (FA) revealed supratemporal bleeding-blocked fluorescence, concomitant capillary non-perfusion zone, and extensive dye leakage from vessels and the optic disc (Figures 1G,H). Positive anterior chamber flare and cells, together with retinal exudation and macular edema, also indicated panuveitis.
The patient has a history of intermittent and recurrent fever, skin rash (Figure 1C), and aphthous ulcer from 5 years ago. Tests from other hospitals implicated hypogammaglobulinemia with immune globulin (Ig) G 4.520 g/l, IgA 4.632 g/l, and IgM 0.134 g/l. One month ago, the patient visited a neurology clinic with complaints of dizziness, nausea, vomiting, totter, and slurred speech. Magnetic resonance imaging (MRI) revealed lacunar infarction in the right basal ganglia and the lateral ventricle (Figure 1E). Activated partial thromboplastin time (APTT) was mildly prolonged to 50.3 s. CBC showed elevated neutrophil percentage (NEU%) of 88.1% and decreased lymphocyte (LYM#) of 0.45 x 109/L. Hepatorenal function, cranial angiography, carotid and cardiac ultrasound, and dynamic electrocardiogram were uneventful. Because of self-claimed allergy to aspirin, patient was administered clopidogrel and atorvastatin once a day.
With consultation from neurologists and rheumatologists, the following laboratory investigations were carried out: raised C reactive protein (CRP) of 41.2 mg/l, homocysteine of 16.0 mumol/l, decreased CD25 + lymphocyte of 4.06%, helper T-cell of 3.70%, IgG of 4.3 g/l, IgA of 0.48 g/l, and IgM of <0.25 g/l, and microcytic hypochromic anemia was noticed. Other workups, including urine routines, erythrocyte sedimentation rate (ESR), coagulation function, streptolysin O, rheumatoid factor, antiphospholipid antibodies, antineutrophilic cytoplasmic antibodies, antinuclear antibodies, protein C, protein S, Toxoplasma gondii, rubella, herpes simplex, hepatitis B, hepatitis C, Epstein-Barr virus, HIV, cytomegalovirus, Treponema pallidum, and Mycobacterium tuberculosis, were negative. Anterior chamber paracentesis for high-throughput sequencing did not find any pathogenic microorganisms.
Oral methylprednisolone 24 mg/day was started and tapered off within 3 months, together with three times of periocular injection of 20 mg methylprednisolone. Topical fluorometholone (0.1%) and pranoprofen (0.1%) eye drops four times per day were administered. Carteolol (2%) and brinzolamide (1%) eye drops were used to alleviate IOP. Focal retinal photocoagulation and intravitreal injection of ranibizumab (0.5 mg/50 mul) were given to control fundus hemorrhage. One week after treatment, KP and aqueous flare and cells disappeared and the IOP decreased back to normal. One month later, the macular edema was resolved in the right eye (Figure 2D). White sheathing of the vessels alleviated (Figure 2A). Two months later, a fundus photo showed full resolution of vascular sheathing (Figure 2B) and visual acuity improved to 20/133. Retinal hemorrhage was absorbed at 5 months follow-up (Figure 2C).
However, the patient came to our emergency room (ER) with a complaint of left limb weakness and deterioration of slurred speech for 3 days, 5 months after being diagnosed of FBA. APTT was prolonged by 47.8 s, F XI and F XII activity was decreased to 66.7% (reference range: 70-120%) and 6.6% (reference range: 70-150%), respectively. MRI confirmed fresh lacunar infarction in the right pons (Figure 1F). The patient was diagnosed with patent foramen ovale (PFO) by contrast echocardiography of the right heart during admission and underwent a catheter-based closure surgery then. One month later, our patient was admitted in the emergency department of another center, complaining aggravation of left limb weakness and slurred speech. Due to the recent cardiac surgical implantation, cranial computerized tomography (CT) was adopted instead of MRI, which implicated low-density focus of the right pons. Considering deteriorating strokes under clopidogrel and atorvastatin intervention, 1.5-2.25 mg oral warfarin was given to prevent cryptogenic stroke, with international normalized ratio (INR) maintained between 2 and 3. One month later, the patient appeared with progressive dizziness and tinnitus and was diagnosed of sudden hearing loss of the left ear. Then, the patient presented with diplopia associated with palsy of the left trochlear nerve. Given the patient's complicated multiple disorders, whole exome sequencing (WES) was adopted to explore the possible pathogenesis. Several deletion mutations of ADA2 (homozygous deletion of exon 7 and heterozygous deletion of exons 2-6 and 8-10) and heterozygous mutation of F12 (p.K365Qfs*69) were recognized. Subsequent laboratory test demonstrated a total ADA activity of 2.2 U/l and an ADA2 activity of 0 U/l. Anti-TNF therapy was then started by weekly injection of 50 mg etanercept. Meanwhile, the patient received intravenous immunoglobulin according to monitoring. The brief timeline of our case is described in Figure 3. | autoinflammatory and autoimmune diseases, blood coagulation factor xii, deficiency of adenosine deaminase 2, frosted branch angiitis, retina vascular disorder | Not supported with pagination yet | null |
PMC8484865_01 | Female | 2 | A 2-year-old Japanese female with no history suggestive of any immune system or metabolic disorder visited our institute before the coronavirus disease (COVID-19) outbreak. She presented with fever 2 days before admission (day 1) and manifested cervical lymphadenopathy on day 2. On day 3, it was observed that she had a persistently high fever, numerous coalescent red spots on the trunk and limbs, peripheral edema, conjunctival injection, and a strawberry tongue. The initial laboratory evaluation showed leukocytosis (24,200/muL) and an elevated C-reactive protein (CRP) level (11.6 mg/dL). A diagnosis of KD was made, and she was admitted to our hospital where she was treated with intravenous immunoglobulin (IVIG), a dose of 2 g/kg/day, and oral aspirin. The recrudescent fever required another IVIG treatment (2 g/kg/day) on day 7. A high fever recurred on day 11, but no other clinical features of KD were present. Laboratory data revealed a decreased white blood cell (WBC) count (6,610 /muL) and CRP level (0.70 mg/dL).
On day 12, the patient experienced episodes of vomiting and a cluster of generalized tonic-clonic seizures without recovery of consciousness. The seizures resolved with multiple intravenous infusions of midazolam. The patient's body temperature was 40.1 C, and her blood pressure 107/58 mmHg. She did not have any upper respiratory tract symptoms or diarrhoea. Laboratory data showed a normal WBC count (7,620/muL), moderately increased CRP level (3.24 mg/dL), and mild hyponatremia (131 mEq/L). Serum glucose was 173 mg/dL, and alanine transaminase and aspartate aminotransferase levels were elevated at 110 and 185 IU/L, respectively. Cerebrospinal fluid (CSF) analysis did not show pleocytosis, but the protein level was elevated to 90 mg/dL. Blood and CSF culture were negative. The FilmArray Meningitis and Encephalitis Panel (BioFire Diagnostics/Biomerieux, Salt Lake City, Utah), a pathogen-specific polymerase chain reaction testing of CSF capable of simultaneously detecting 6 bacteria, 7 viruses and a yeast, yielded negative results. Cranial computed tomography (CT) revealed no abnormalities. After convulsive status epilepticus, electroencephalography (EEG) showed a high-amplitude diffuse slowing background without ictal activity. During the interictal period, the patient could communicate with her parents and drink water.
Several convulsive seizures were observed on day 13. Moreover, the patient's mental status started to worsen and she became lethargic. MRI T2-weighted images (T2WI) showed bilateral symmetrical hyperintensities in the cerebral white matter and thalamus. The white matter lesions were observed predominantly in the subcortical white matter and sparsely in the internal capsule and corpus callosum (Figure 1). Linear radial hyperintensities parallel to the cerebral vessels of the periventricular space were also observed (Figure 1C). The white matter lesions were isointense on a T1-weighted image (T1WI), and there were hyperintense signals on fluid-attenuated inversion recovery (Figures 2A,B). The lesions showed no gadolinium enhancement (Figure 2C). The cerebral white matter displayed iso-intensity on diffusion-weighted images (Figure 2D), with high intensity on the apparent diffusion coefficient map (Figure 2E). These findings suggested vasogenic edema. Susceptibility-weighted images did not show any hemorrhages.
Eight hours after the MRI examination, the patient had abrupt cessation of spontaneous breathing that required intratracheal intubation and mechanical ventilation. Her pupillary light reflexes were absent, and her EEG showed diffuse low activity. Head CT demonstrated diffuse swelling of the cerebrum, cerebellum, and brainstem, with sulcal and cisternal effacement, suggesting brain herniation (Figure 2F). We diagnosed the patient with acute encephalitis related to KD based on the clinical manifestations, such as high fever, a cluster of seizures, vomiting, altered mental status, and neuroradiological abnormalities. We started intensive treatment with intravenous methylprednisolone, plasma exchange, mannitol, inotropic agents, and target temperature monitoring, but these were not effective for the brain swelling. The patient developed neurogenic pulmonary edema and central diabetes insipidus. The background activity on the EEG remained severely depressed, and the brainstem reflexes did not recover, and the patient died due to septic shock on day 41. Echocardiography was performed on days 3, 5, 6, 8, 11, and 13, and it did not show any coronary artery lesions. The additional laboratory tests for serum anti-myelin oligodendrocyte glycoprotein antibody and anti-N-methyl-D-aspartate receptor antibody in the CSF were negative. | kawasaki disease, acute brain swelling, acute encephalitis, acute encephalopathy, acute fulminant cerebral edema, case report, perivascular lesions, vasogenic edema | Not supported with pagination yet | null |
PMC5259670_01 | Male | 73 | A 73-year-old Caucasian male with past medical history of Pott's disease as a child, osteoarthritis of the right knee, and a right Baker's cyst presented to the Emergency department with complaints of worsening leg pain for two days and confusion for one day. At presentation, the patient was tachycardic. All other vital signs were within normal limits. His review of systems was remarkable for chronic knee pain for the last two years and right leg pain for the last three months and decreased appetite and weight loss over the course of four months. On physical examination, the patient was in moderate distress. He was alert and oriented to time, place, and person but had poor concentration. On musculoskeletal exam there was a diffuse area of swelling on the right lower extremity which was extremely tender to touch with minimal warmth and erythema. This was located posteromedially involving the calf and medial aspect of the leg. Knee joint was nonswollen, nontender, and without any erythema or warmth (Figure 1). Laboratory data revealed leucocyte count 14.6 k/cumm, Hemoglobin 10.2 mg/dL, and hematocrit 30.4%. HIV testing was negative. His metabolic panel showed hyponatremia, 124 mEq, but was otherwise unremarkable. Leg MRI was performed. This showed a large heterogeneous fluid collection involving the subcutaneous soft tissues of the posterior medial right lower extremity measuring approximately 21.1 x 7.2 x 8.5 cm with marked mass effect on the gastrocnemius muscle. Heterogeneity of the distal femur including the femoral condyles, tibial plateau, and proximal fibular head was also visualized suggesting possible osteomyelitis as well (Figure 2). The patient underwent surgical resection of the left Baker's cyst. Intraoperative findings included a thick, boggy synovium with dull and dusky appearing cartilage. Tissue and fluid stain revealed acid fast bacillus (AFB) in one of two samples which was later identified as MTB. On pathologic section, a granulomatous reaction with giant cells was present. The patient was placed in airborne isolation and a workup for dissemination was pursued. Respiratory samples also had AFB identified on initial acid fast staining. Chest CT showed multiple apical cavitary lesions and nodular infiltrates with tree in bud opacities in the apices and right lower lobe. Acute kyphotic deformity of the upper thoracic spine with loss of vertebral body height, vertebral body fusion, posterior element fusion, and kyphosis consistent with the patient's known history of Potts disease was also seen in chest CT. Due to worsening mental status, a diagnostic lumbar puncture was also performed. CSF analysis showed 1 red blood cell/UL, 151 white blood cells/UL, 64% neutrophils, protein 1668 mg/dL, and glucose of 8 mg/dL. CSF polymerase chain reaction tested positive for MTB. Testing for HIV, Hepatitis B, and C was negative. Treatment with rifampicin, ethambutol, isoniazid, pyrazinamide, and dexamethasone was started. During the course of therapy, patient developed pneumonia and hypoxic respiratory failure requiring intubation. Family chose to proceed with comfort measures only and the patient passed away due to septic shock. | null | Not supported with pagination yet | null |
PMC10205527_01 | Female | 24 | We present a case of a 24-year-old woman who works as a teacher on an indigenous reservation in a rural area. She often shared meals with the community, including dairy products like unpasteurized cheeses produced in the region. The patient has actively participated in animal sterilization events for dogs and cats and has been exposed to outdoor poultry. She has two children and no significant medical history, including hypertension or diabetes. The patient has never smoked or used drugs and has no significant sick contact, known TB exposure or recent travel to other endemic areas.
The patient consulted the emergency department due to a six-month history of constitutional and gastrointestinal symptoms, including bloating and diarrhea, along with a critical weight loss (15 kg), low-grade fever, nocturnal diaphoresis, gradual onset of ascites, and abdominal pain. The patient had previously consulted a local medical center, receiving treatment for gastrointestinal and urinary infections with frequent courses of antibiotics without clinical improvement. During the consultation, the patient reported no headache, cough, dyspnea, chest or joint pain, urinary symptoms, or menstrual cycle irregularities. Her vital signs were the following: Blood pressure of 100/70 mm Hg, heart rate of 90 beats/minute, respiratory rate of 14 breaths/minute, and temperature of 37.5 C .
On physical examination, she appeared thin and pale, and tolerated decubitus. The most significant finding was abdominal distension with a positive ascitic wave without collateral circulation, telangiectasias, or pain upon palpation. The moist mucosa showed no signs of ulcers; there was no jaundice, no jugular vein distention, no neck masses, a rhythmic heart without murmurs, and normal lung sounds. The extremities had no edema present, and the neurological exam was unremarkable.
The patient underwent initial laboratory tests outlined in (Table 1). The PPD skin test was negative, and the results of autoimmune testing showed some alterations and an elevated CA-125 level. A chest X-ray was conducted but yielded no significant findings. However, an abdominal computed tomography (CT) scan revealed thickening of the parametrium, parietal peritoneum, omentum, gastric cavity, intestinal lining, and intraperitoneal liquid with intermediate density in all compartments (Fig. 1). To further analyze the ascitic fluid, abdominal paracentesis was performed. The fluid was predominantly lymphocytic and had a serum ascites albumin gradient (SAAG) index of 0.3 (Table 2), which ruled out portal hypertension. Therefore, additional investigations were conducted to exclude the possibility of malignant pathologies, infectious, and inflammatory diseases.
The patient was evaluated by a gynecologist who performed a transvaginal ultrasound to investigate the possibility of gynecologic malignancies but found no abnormalities. Similarly, to rule out gastrointestinal malignancies, the patient underwent both endoscopy and colonoscopy, which were unremarkable. On the fourth day of hospitalization, a diagnostic laparoscopy revealed a friable peritoneum with numerous miliary-like micronodules covering the entire peritoneal and pelvic cavity (Fig. 2). Three days later, the pathology report revealed fibroconnective tissue with multiple confluent granulomas, high histiocytic cellularity and multinucleated giant cells surrounded by a lymphoplasmacytic inflammatory infiltrate, some of which contained central necrosis (Fig. 3).
Under the suspicion of peritoneal tuberculosis (PTB), the patient was started on anti-tuberculosis therapy eight days after admission. She began a six-month regimen of isoniazid, rifampicin, pyrazinamide, and ethambutol. The patient showed progressive improvement with the current treatment. A positive adenosine deaminase (ADA) test result of the peritoneal fluid was reported the following day and supported the diagnosis of PTB. Despite negative microbiological culture for bacteria of ascites fluid, gram stain, Auramine-rhodamine stain, and GeneXpert MTB/RIF, peritoneal tuberculosis was confirmed four weeks after the diagnostic laparoscopy through a positive culture for mycobacteria in the peritoneal tissue.
The patient was discharged 15 days after admission with a noticeable improvement in the initial symptoms. She has been followed up in the infectious disease clinic for a year after finishing the antituberculous treatment. She reported weight gain, adequate oral tolerance, and no new episodes of diarrhea. | ascites, ascitic fluid, case report, peritoneal tuberculosis, tuberculosis, tuberculosis-gastrointestinal | Not supported with pagination yet | null |
PMC6196681_01 | Female | 32 | A 32-year-old woman presented with generalized colicky abdominal pain, bilious vomiting and constipation of 1-day duration. She had no history of abdominal surgery or constitutional symptoms of chronic illness such as tuberculosis, inflammatory bowel disease or malignancy. She had been married for more than 10 years and has one child. Her vital signs were normal. Complete blood count, liver and renal function tests, erythrocyte sedimentation rate and C-reactive protein levels were normal. Abdominal examination revealed diffuse distention with hyperactive bowel sounds; there were no hernias or signs of peritonitis. The clinical impression and plain abdominal X-ray findings were consistent with subacute SBO. Initially, the patient improved on conservative management of nil by mouth, intravenous (IV) fluids and nasogastric tube (NGT) decompression. Abdominal computerized tomography scan with water-soluble contrast through NGT revealed dilated loops of small bowel, left hydrosalpinx and contrast reaching the colon. The patient underwent laparoscopic intervention because of persistence of her symptoms and to establish the underlying cause. Laparoscopic findings were multiple small bowel adhesions (mainly ileal), "violin string" adhesions between the liver and anterior abdominal wall, free peritoneal fluid and left hydrosalpinx [Figure 1]. Laparoscopic adhesiolysis was performed using endoshears and diathermy [Figure 1]. Swab samples were initially taken from the peritoneal fluid and later from the uterine cervix to test for Neisseria gonorrhoeae and Chlamydia trachomatis. Swabs and serology for both organisms were negative. Gynecological consultation was sought, and no antibiotics were recommended because the patient did not have active PID. The patient had an uneventful recovery and was discharged 5 days after surgery. Two months later, she remained symptom-free when seen at the outpatient clinic. | fitz-hugh–curtis syndrome, laparoscopy, pelvic inflammatory disease, small bowel obstruction | Not supported with pagination yet | null |
PMC6206551_01 | Male | 48 | A 48-year-old Indian male with no chronic medical illness in the past admitted to emergency department with history of fever, headache, dry cough, and generalized body pain for 4 days and vomiting for one day. No abdominal pain, SOB, chest pain, joint pain, skin rash, recent travel, or exposure to sick person and no significant family history were reported. Patient denied alcohol consumption or tobacco smoking. On physical examination, the patient was well built and nourished; he was icteric, conscious, and oriented to time, place, and person. Vitals were as follows: temperature: afebrile, 35.9 C; heart rate: 94/minute; respiratory rate: 20/minute; blood pressure: 121/81 mmHg; and SpO2: 98% in room air.
Systemic examination showed normal neurological findings except meningeal signs. Other systems were unremarkable. Initial investigations showed hemoglobin and platelets were normal. White blood cell (WBC) count was 12.6 x 103/microliter (normal: 4 x 103/microliter-10 x 103/microliter) with 92% neutrophils. Serum creatinine was 146 micromol per liter (normal: 64 to 110 micromol per liter), urea was 11 mmol per liter (normal: 3.2 mmol per liter to 7.4 mmol per liter), and serum electrolytes were normal. Alanine aminotransferase (ALT) was 56 units per liter (normal: 0 units per liter to 30 units per liter), aspartate aminotransferase (AST) was 38 units per liter (normal: 0 units per liter to 31 units per liter), alkaline phosphatase (ALP) was 96 units per liter (normal: 40 units per liter to 150 units per liter), albumin was 33 g per liter (normal: 35 g/L to 50 g/L), total bilirubin was 68 micromol per liter (normal: 3.4 to 20.5 micromol per liter), direct bilirubin was 34 micromol per liter (normal: 0 to 8.6 micromol per liter), C-reactive protein (CRP) was 495 mg per liter (normal: 0 to 5 mg/L), procalcitonin (PCTN) was 11 ng per milliliter (normal: 0 to 2 ng/mL), and chest X-ray was normal. His conscious level deteriorated soon after hospital admission, and the Glasgow Coma Scale (GCS) dropped from 15/15 to 12/15. Meningitis is suspected, and antibiotics were started after lumbar puncture (LP) and computerized tomography (CT) head. Initial empirical intravenous (IV) antibiotics were started: ceftriaxone, vancomycin, and acyclovir along with dexamethasone. CT head was normal, and cerebrospinal fluid (CSF) analysis showed WBC 145 per microliter (normal: 0 to 5/microL) with 96% neutrophils, protein 4.55 grams per liter (normal: 0.15 to 0.45 g/L), and Glu <0.3 mmol per liter; CSF viral panel showed Epstein-Barr virus (EBV) PCR was 326 International Units per mL, acid-fast bacilli staining was negative, and tuberculosis PCR was negative. Both CSF and blood culture showed Streptococcus pneumonia which was sensitive to ceftriaxone. So acyclovir and vancomycin were stopped, dexamethasone was given for a total of 3 days, and IV ceftriaxone 2 g every 12 hours was continued; later, the patient's condition improved and oriented to person but not oriented to time and place. There was no focal neurological deficit. However, he developed hard of hearing and though fever pattern has improved, having fever spike on and off. Repeat blood cultures were negative, CRP improved from >500 mg/L to 93 mg/L, PCTN decreased from 11 ng/mL to 0.41 ng/mL, and serum creatinine level normalized. However, liver enzymes persisted to rise (both transaminases and alkaline phosphatase), and white blood cell persisted to be in the range of 13-15. Due to presence of persistence of symptoms, magnetic resonance image (MRI) brain and repeat LP were done to rule out any complication of the disease. MRI brain (Figures 1 and 2) showed meningoencephalitis, vasculitis, and extradural fluid collection. There was right-sided fluid in the mastoid cavity without bone destruction. ENT was consulted and advised for medical management. As the patient is confused, hearing assessment could not be done properly. Repeat LP showed CSF WBC 11/microL, neutrophils 70%, lymphocytes 29%, protein 1.11 g/L, EBV PCR was negative, and CSF culture was negative. Dexamethasone was restarted with continuation of IV ceftriaxone for a total of 6 weeks as Streptococcus pneumoniae meningitis is complicated by infective vasculitis, mastoiditis, and subdural collection. Repeat MRI brain (Figure 3) showed significant improvement in leptomeningeal enhancement and resolution of epidural collection; however, there was a new communicating hydrocephalus. After completion of his IV ceftriaxone, the patient was repatriated to his home country. Although his condition improved on above treatment, he was discharged with mild disorientation to time and person. | null | Not supported with pagination yet | null |
PMC3420757_01 | Female | 22 | A 22-year-old woman presented with 6-8 weeks of dysphagia. She also reported a choking sensation while eating food and brushing her teeth. She had a known history of uvular elongation, without change in size, for several years. She also complained of globus sensation, frequent throat clearing, dry cough, excess mucus, and heartburn. Examination revealed a pedicled lesion, approximately 1.5 cm in length, extending from the inferior tip of her uvula (Figure 1(a)).
The lesion was completely excised from its connection to the uvula under general anesthesia with electrocautery (Figures 1(b) and 1(c)). Pathology revealed characteristic findings of a squamous papilloma, including multiple squamous lined papillary fronds containing fibrovascular cores (Figures 2(a) and 2(b)). The patient's postoperative course was unremarkable. Although she remains now on pharmacologic treatment for acid reflux, she reported complete resolution of all symptoms. | null | Not supported with pagination yet | null |
PMC10164848_01 | Female | 11 | A previously healthy 11-year-old female presented with abdominal pain for one month and a fever for 15 days. Her medical history was significant for unexplained weight loss over the past month. She was referred from a tertiary care hospital with reported hepatomegaly (13 cm liver span), moderate splenomegaly (16.6 cm), and right-sided minimal pleural effusion on ultrasonography.
On arrival at our hospital, her temperature was 98 F. She was tachycardic with pulse being 140 bpm, and her arterial blood pressure was 108/66 mmHg. Our physical examination referred to epigastric tenderness, abdominal distension, and hepatosplenomegaly with decreased breath sounds on the right side of the chest. Our laboratory results reported pancytopenia (Hemoglobin 7.9 mg/dl, Platelets 79 x 10E9/L, and WBC 5.5 x 10E9/L). Her peripheral smear showed mild microcytic, hypochromic, anisocytosis, elliptocytes, polychromasia, and dimorphic picture of RBCs and few macrophages. Laboratory testing was otherwise significant for Direct Bilirubin (0.4 mg/dl) and Hypoalbuminemia (2.34 g/dl). Upper GI Endoscopy was completely normal. Anti-tissue transglutaminase Ig A levels, ANA levels, serum ceruloplasmin levels, and 24-h urinary copper levels were all within range. CT scan of the abdomen with contrast showed hepatosplenomegaly, mild ascites, multiple enlarged lymph nodes, and a focal area of narrowing in the sigmoid colon. The right middle lung lobe showed patchy ground-glass haziness with few fibrotic nodules, and mild to moderate right-sided pleural effusion was noted (Figure 1). Due to her abdominal pain, we started her course for IV analgesics, but her condition did not improve. Initial infectious workup was unrevealing, with negative malaria, human deficiency virus, Tuberculin skin test, and negative dengue serology. Thoracentesis showed exudative pleural effusion with inflammatory cells rich in lymphocytes, few mesothelial cells, and foamy macrophages; no malignant cells were seen in the fluid. Pleural fluid direct microscopic examination revealed no presence of tuberculosis bacilli. She became very irritable during the hospital stay and had an episode of fit/delirium/psychosis. In addition to the attack, brain magnetic resonance imaging was done, which reported no abnormality except for minimal meningeal enhancement along the right parietal lobe (Figure 2). Lumbar Puncture was advised, which was refused by the attendant. Systemic lupus erythematosus was suspected due to the occurrence of acute psychotic events, but the anti-ds antibody came out negative. However, Brucella antibody titers, Abortus antibody, and Melitensis antibody were >1:320; this explained the presence of Brucella melitensis. Initially, she was started on Injection Meropenem 1 g for six days. After positive titers, Syrup Doxycycline (50 mg/5 ml) and Syrup Rifampicin (2 g/100 ml) were prescribed for six weeks. The patient was followed up in OPD, and symptoms were improved. Rifampicin 8 ml was further continued for 15 days. | brucellosis, case report, infectious disease, pediatrics, zoonotic disease | Not supported with pagination yet | null |
PMC5574471_01 | Female | 19 | A 19-year-old US-born college student with a past medical history significant for ulcerative colitis treated with infliximab for the past 3 years and negative tuberculin skin test at initiation of TNFA was evaluated for a 3-month history of fever, night sweats, weight loss, productive cough, and abdominal pain. Several weeks prior to evaluation, she was treated for community-acquired pneumonia with azithromycin without symptomatic improvement. She was additionally treated with a short course of ciprofloxacin and prednisone for possible ulcerative colitis exacerbation without improvement. She had no known TB exposure including prior travel to endemic TB areas.
Physical examination revealed an afebrile woman in mild distress. Vital signs were notable for mild hypoxia (Spo2 = 92% on room air). She had no palpable lymphadenopathy. Bilateral rhonchi were present on pulmonary auscultation. Remainder of physical examination was unremarkable.
Laboratory evaluation revealed a normal complete blood count and inflammatory markers. Human immunodeficiency virus (HIV) testing was negative. QuantiFERON-TB Gold in-Tube (QFT) test was positive (2.62 IU/mL). Transbronchial lung biopsy and bronchoalveolar lavage showed acid-fast bacilli, and subsequent cultures grew pan-sensitive MTB. Computed tomography of the chest, abdomen, and pelvis revealed miliary pulmonary pattern, patchy nodular infiltrates, and mediastinal lymphadenopathy with peritoneal and omental involvement (Figure 1). She did well after completion of 6 months of anti-TB therapy.
In addition to the clinical QFT test, peripheral blood mononuclear cells (PBMCs) were analyzed by FC as part of a research study. This research study was approved by Mayo Clinic Institutional Review Board (Mayo IRB number 09-003253 00). Peripheral blood mononuclear cells were isolated by Ficoll-Paque separation from 40 mL of heparinized blood within 1 hour of collection and cryopreserved in liquid nitrogen until stimulation. Multiparameter antigen stimulation with costimulatory antibodies (MTB-purified protein derivatives (PPD), region of difference 1 (RD1) peptide antigen [ESAT-6/CFP-10 peptide mix or specific MTB antigens], positive and negative controls) was completed. The PBMC sample and antigens were incubated for 48 hours at 37 C and then stained with fluorescent dye-conjugated anti-CD3, anti-CD4, anti-CD8, anti-CD25, and anti-CD134 antibodies and isotype controls. About 2 x 105 cells were analyzed by fluorescence-activated cell sorting (FACS) (BD FACSCanto) and gated using FlowJo software and Kaluza FC software. Detailed methods have been reported previously.
CD25+CD134+ co-expression was detected on 0.34% and 0.84% of RD1 peptide and PPD-stimulated CD3+CD4+ T cells, respectively (Figure 2). In addition, upregulation of CD25+CD134+ was present on 0.26% and 0.59% of RD1 peptide and PPD-stimulated CD3+CD8+ T cells, respectively. These results were in the range of untreated LTBI associated with an increased risk of TB reactivation, as previously described, and suggest possible active TB infection in an immunosuppressed and symptomatic patient. | tumor necrosis factor alpha antagonists, active tuberculosis, flow cytometry, inflammatory bowel disease | Not supported with pagination yet | null |
PMC9084234_01 | Male | 40 | We describe a 40-year-old male patient with relapsed B-common ALL who developed Sars-CoV2 prior to treatment initiation. In December 2020, the patient with a Klinefelter syndrome and a previous B-common ALL treated with the pediatric-inspired trial NILG ALL10/07 in 2015, off-treatment since 2015, presented pancytopenia, fever, and dyspnea. At Viterbo hospital, peripheral blood morphological examination showed the presence of blasts with a lymphoid habitus. After a negative Sars-CoV2 nasopharyngeal swab, the patient was admitted to our Center in Rome. Laboratory tests confirmed the pancytopenia (Hb 10.3 g/dL, WBC 0.64 x 109/l, Plts 43 x 109/l) and fluorocytometric analysis revealed the presence of 77% CD10+, CD22+, CD45+, CD19+, CD34+, CD123+, CDc79a+, TdT+ blasts, consistent with a diagnosis of relapsed B-common ALL. The molecular profile was negative for the major molecular aberrations (i.e., BCR/ABL1, E2A/PBX1, KMT2A-r).
At relapse, a chest C.T. scan documented a pulmonary embolism, and a Doppler ultrasound showed a saphenous thrombosis of the left leg with a concomitant subcutaneous abscess with cellulitis, extended from the inguinal region to the femoral-popliteal area, which required fondaparinux administration and antibiotic treatment (piperacillin/tazobactam, metronidazole, and daptomycin). In addition, the patient started methylprednisolone, 60 mg/day, but on 1 January 2021, the Sars-CoV2 nasopharyngeal swab proved positive (later confirmed also on bronchoalveolar lavage (BAL), and he was therefore transferred to a COVID-19 ward at our hospital.
The patient did not receive COVID-19 vaccination since no vaccination was available at the time of relapse.
Our ALL strategy contemplated 2 cycles of inotuzumab (0.8 mg/m2 D1, 0.5 mg/m2 D8 and D15) and an allogeneic transplant (allo-SCT), depending on donor availability. Since the patient was asymptomatic for COVID-19 infection and the C.T. scan was negative, on 5 January 2021, the first dose of inotuzumab was administered, followed by the anti-viral drug remdesivir as prophylaxis for ten days (loading dose 250 mg/day, then 100 mg/day) starting on day +1 after inotuzumab. However, on day 9, the clinical conditions worsened, with an acute respiratory syndrome requiring oxygen support; chest C.T. showed bilateral multilobar ground-glass opacities (Figure 1A). By day 15, after eight days of neutropenia, an improvement in the radiological findings and respiratory symptoms was documented. Since a bone marrow aspirate showed a partial response (30% of blasts), on 22 January (day 17), the second dose of inotuzumab (0.5 mg/m2) was administered. As already observed after the first dose, the respiratory syndrome worsened (Figure 1B) but again improved with oxygen and steroids support without non-invasive ventilation; fondaparinux was already ongoing for the pre-existing thrombosis. This condition lasted until day +50 when it gradually improved. On day +49, we documented the presence of an antibody response to Sars-Cov2 (33.6 U/ml, cut-off >1U/ml), but the Sars-Cov2 nasopharyngeal swab was persistently positive (Figure 2); thus, convalescent plasma (CP, 200 ml/day continuous infusion for three days) was administered, without complications.
On day +67, a bone marrow aspirate revealed a complete hematological remission with a minimal residual disease of 1% by flow cytometry, and on day +75, after 20 days from the C.P. infusion, the patient had his first Sars-CoV2 PCR negative result (Figure 2).
On day +84, the patient was dismissed in hematological remission, with a 1% persistence of leukemic cells by flow cytometry, a negative swab for Sars-Cov2, and complete recovery of COVID19-related pneumonia. Meanwhile, there was a slight worsening of the lower left limb cellulitis, despite the continuous administration of antimicrobic treatment. In addition, MRI was suggestive of osteomyelitis; thus, the patient was kept on oral rifampicin and levofloxacin for an additional month.
He returned to Sicily, where he received the second cycle of inotuzumab and two doses of I.T. methotrexate, achieving MRD negativity (by flow and IG/TR). The sars-COv2 vaccine was not administered to the patient since the patient was going to be allografted shortly after and also because, as already mentioned, he did indeed developed antibodies. Therefore, after a conditioning regimen with TBI and cyclophosphamide, on 9 July 2021, the patient received a MUD allo-SCT. He presented a mild vein occlusive disease (VOD), treated with defibrotide. Stem cell engraftment was on day 17. The patient is currently in good clinical condition, with a normal blood count and without graft signs.
To date, few cases are reporting the management of ALL during COVID-19 infection. Treating ALL patients with a concomitant COVID 19 infection is a challenge, since a therapeutic delay might negatively impact prognosis. Different reasons drove our therapeutic choice: the presence of a novel and poorly known viral disease, the initial patient's management in a non-hematology department, and his wish to continue treatment in Sicily. Considering these aspects, we deemed that inotuzumab ozogamicin was the most appropriate choice since it is more manageable than blinatumomab. Indeed, inotuzumab administration does not require a continuous infusion, which may interfere with remdevisir infusion.
Furthermore, at variance from blinatumomab, whose most frequent side effect is represented by the cytokine release syndrome, which can confound and worsen the clinical picture of COVID-19, inotuzumab is in this respect is more manageable; lastly, inozutumab is a more potent compound in treating ALL with a high burden of disease. Finally, inotuzumab is more myelosuppressive than blinatumomab but less than systemic chemotherapy. This effect may have led to an exacerbation of the respiratory symptoms related to COVID19 pneumonia after each dose.
The optimal COVID-19 pneumonia treatment remains unclear and particularly delicate in onco-hematologic patients. Remdesivir is a nucleotide analog prodrug that inhibits viral RNA replication. Recent guidelines recommend remdesivir in the presence of a severe infection. A multicenter randomized trial of remdesivir versus placebo for COVID-19+ patients demonstrated that patients treated with remdesivir have a shorter median time to recovery than the placebo group (10 vs. 15 days) and a lower mortality rate (6.7% vs. 11.9%). However, the optimal duration of treatment with remdesivir is not well-established. In our COVID19 center, remdesivir is used for five days in immunocompetent patients. Supported by other studies, we opted to use remdesivir for ten days in our patient - given the primary hematologic neoplasm - as a bridge between the first and second dose of inotuzumab. This choice appeared appropriate since its use improved the respiratory distress that occurred after the administration of inotuzumab.
Nevertheless, the patient remained persistently positive in the nasopharyngeal swab for Sars Cov2. A turning point was the use of C.P., a passive immunization method previously applied to other viral infections. The neutral antibodies (NAbs) against Sars-Cov2 inhibit the entry and viral amplification and activate different pathways (complement activation, antibody-dependent cellular cytotoxicity, and phagocytosis).
It was reported that C.P. might reduce mortality and improve symptoms in the elderly with COVID-19, though with disappointing results in the non-onco-hematologic population. Data on the use of C.P. in COVID-19 patients are increasing, but the experience in immunocompromised patients is very limited. Immunocompromised patients are unable to develop an appropriate humoral immune response to SARS-CoV-2 and are ideal candidates for passive immunization. Tremblay and colleagues studied 24 patients treated with C.P. with cancer, including 14 with hematological malignancies: of the latter, eight were discharged, one was mechanically ventilated, and five died. Heuso et al. treated 17 patients with CP (15 with hematological diseases). All but one patient showed a clinical improvement, and 9/17 documented a virus clearance. Britains and collaborators studied five onco-hematologic patients with COVID-19 treated with C.P. All patients developed NAbs, and all but one witnessed a clinical improvement. However, there is not yet a consensus regarding the dose and timing; results of studies with a larger number of patients are awaited (NCT04393727).
Unexpectedly, our patient developed antibodies against Sars-Cov2 before C.P. administration: despite the administration of inotuzumab weakening the patient's immune system, he possibly presented a subset of lymphocytes capable of producing antibodies.
Our experience demonstrates the complexity in the care of ALL patients and Sars-Cov2 infection. At present, the patient has recovered from COVID-19 and is at five months from allogeneic transplant in complete molecular remission and good clinical conditions, clearly showing that treatment must be continued, if feasible, and that a multidisciplinary approach is required for the optimal management of these cases. | acute lymphoblastic leukemia, covid-19, convalescent plasma, inotuzumab, remdesevir | Not supported with pagination yet | null |
PMC5485283_01 | Male | 53 | A 53-year-old African American male was brought by Emergency Medical Services (EMS) to the Emergency Room (ER) of an urban, public teaching hospital, due to threatening behavior, irritability, and an inability to care for himself. During the initial psychiatric consultation in the ER, the patient was hyperverbal with pressured speech and a tangential thought process. His mood was elevated, and his affect was labile with sudden and inappropriate bouts of tearfulness. He endorsed decreased need for sleep over the past few days and paranoid and persecutory delusions regarding strange noises around his apartment and his brother stealing money from his father. Per the EMS report, the patient was also emailing and texting neighbors paranoid and threatening messages, which resulted in multiple crisis hotline calls and the patient being brought to the hospital.
His past medical history was significant for a depressive episode treated successfully 25 years ago with sertraline and TBI five months prior to presentation. After the injury, he was followed by an outpatient neurologist for postconcussive syndrome. A brain MRI was ordered three months after TBI, which showed signs of mild white matter small vessel ischemic changes, but no other significant findings. The initial ER workup included a urinalysis, urine drug screen, complete metabolic panel, and thyroid function, all of which were unremarkable. A noncontrast CT scan of the brain was obtained and was unremarkable. He was admitted for inpatient psychiatric hospitalization. Using Diagnostic and Statistical Manual of Mental Disorders, 5th edition (DSM-5) criteria, he was diagnosed with bipolar disorder due to traumatic brain injury, with manic features.
The patient was started on olanzapine 2.5 mg by mouth at bedtime upon admission. Manic features remained prominent, as he continued to demonstrate decreased need for sleep (three to five hours per night), pressured speech, irritability, emotional dysregulation, and labile affect. He often arose early in the morning and spent multiple hours writing questions for his treatment team. He refused valproate and lithium despite the team's suggestions. Olanzapine was gradually titrated and reached 15 mg on hospital day (HD) 6. Aside from one instance of refusal on HD 5, he was adherent with olanzapine throughout the hospitalization. Olanzapine was well tolerated. He was evaluated with serial clinical rating scales which included the Young Mania Rating Scale (YMRS) and the Clinician-Rated Dimensions of Psychosis Symptom Severity (CRDPSS). On HD 1, the YMRS score was 31 and CRDSS was 9. On HD 6, the YMRS score decreased to 30 and CRDPSS decreased to 7. On HD 7, he displayed significant clinical improvement. He slept throughout the night and on interview no longer had pressured speech. His thought process was logical, linear, and goal-directed, and the psychotic symptoms had also fully abated. On HD 8, the YMRS and CRDPSS scores were 0. He was subsequently discharged from hospital with appropriate outpatient follow-up. | null | Not supported with pagination yet | null |
PMC7476695_01 | Female | 58 | The patient was a 58-year-old woman who presented with progressive low back pain for 3 months before admission. She had no history of hypertension, diabetes mellitus, systemic carcinoma, or congenital tissue abnormalities. On admission, she exhibited marked gait disturbance with low back pain and sciatic nerve pain, which was dominant on her right side. The strength of the right tibialis anterior and extensor hallucis longus muscles was Grade 3-4, and sensory disturbance, including pain as well as thermal and touch sensations, occurred on the bilateral lower thigh. Findings of deep tendon reflexes and pathological reflexes were normal. Blood examination results revealed a tartrate-resistant acid phosphatase (TRACP)5b level of 967 mU/dl and an alkaline phosphatase (ALP) level of 393 ml/dl (Table 1). Initial X-ray and computed tomography (CT) results revealed the presence of an osteolytic lesion involving the L4 vertebra with the abnormal alignment of anterior spondylolisthesis (Fig. 1a and b). Magnetic resonance imaging (MRI) revealed low signal intensity and intermediate-to-high signal intensity areas on T1- and T2-weighted images, respectively. Gadolinium-enhanced axial images revealed that the tumor mass expanded into the extravertebral and intraspinal canal spaces (Fig. 1c-e). Results of CT-guided percutaneous biopsy were consistent with the diagnosis of giant-cell tumor of the spine.
As per the protocol in a previous study, the patient was preoperatively administered 10 courses of denosumab (120 mg at a time) in monthly cycles with initial additional loading doses. Following denosumab therapy, her severe low back pain was resolved and no neurological deficits were noted. Follow-up pre-operative X-ray and CT revealed progression of anterior deviation and an increase in vertebral body mineralization (Fig. 1f and g). MRI revealed a decrease in tumor enhancement and a marked reduction in the tumor size in the retroperitoneum space and intraspinal canal region (Fig. 1h-j). The TRACP5b and ALP levels returned to normal after 3and 10courses of denosumab therapy, respectively (Table 1).
A combined anterior and posterior approach was used to perform the total spondylectomy of the L4 vertebra. First, we exposed the dura mater by en bloclaminectomy and detached the bilateral L4 pedicles using a threaded T-saw. The lamina and pedicles were rigid, and there was no adhesion between the dura mater and tumor section. The lateral aspect of the L4 vertebral body was exposed, and disc resection was performed using the posterior incision approach. Using the anterolateral approach, the vertebral body was resected piece by piece, and the bone in the regenerated cortical GCTB was treated with denosumab. Furthermore, corpectomy of L4 was performed; subsequently, titanium cage reconstruction was performed with posterior L3-L5 stabilization.
Postoperatively, the patient remains pain free without neurological symptoms, and there is no radiologic evidence of tumor recurrence for 1 year after surgery, i.e., following three courses of denosumab administered at 2-month intervals.
Light microscopy revealed numerous multinucleated giant cells in the background of neoplastic mononuclear stromal cells in pre-operative biopsy samples (Fig. 2a). Immunohistochemical staining revealed RANK-positive mononuclear cells and aggregations of strongly cyclooxygenase-2 (COX-2)-positive mononuclear stromal cells (Fig. 2b and c). The microscopic examination of tissues harvested after denosumab therapy revealed a fibrous matrix with no multinucleated giant cells (Fig. 2d). Immunohistochemistry detected RANK-positive cells around the woven bone, although almost all of the RANK and COX-2-positive cells disappeared (Fig. 2e and f). | denosumab, giant-cell tumor, immunohistochemistry | Not supported with pagination yet | null |
PMC8379400_02 | Female | 12 | A 12-year-old female, who was the firstborn of a non-consanguineous marriage, was diagnosed with anotia on the left side, cyanosis, and facial asymmetry at birth. Figure 1 shows absent ear cartilage and external auditory canal on the left side. She was operated for truncus repair with 12mm aortic homograft and PDA ligation was performed at 2 months of life. She was then put on anti-failure drugs. On examination of the cardiovascular system, S2 had a wide split, and ejection systolic murmur was of intensity 3/6 at the lower-left sterna border. The oxygen saturation (SPO2) was 68 % in both the upper extremities. Findings of 2D Echocardiograph suggested of patent ductus arteriosus (PDA), truncus arteriosus type I, ventricular septal defect (VSD), and severe Pulmonary artery hypertension. Ultrasonography disclosed unremarkable study of kidney, ureter and bladder region. The patient grew with normal development and scholastic performance and doing well in school but had restrictions on physical exertion. Moreover, she had moderate growth failure. Upon audiogram, right ear mild conductive hearing loss and left moderately severe conductive hearing loss was found. High-resolution computed tomography (HRCT) of the Petrous Temporal was suggestive of non-visualisation of left external ear (pinna), left auditory canal suggested of agenesis of the left external ear. Both mastoid air cells demonstrated sclerosing and loss of pneumatisation, which was suggestive of bilateral sclerosing mastoiditis. Besides, the course of the left facial nerve was normal.
Patient started to have a deformity of upper back at the age of 10 years, which was progressive and caused mild left shoulder elevation. She did not present with neurological deficits. Her coronal and sagittal balance was good without truncal imbalance. The X-ray of the spine showed kyphoscoliosis as shown in Figure 2. Both left thoracic curve and right proximal thoracic curve were rigid in finding, with a rib hump of grade 3. Magnetic resonance imaging (MRI) of the whole spine showed the congenital fusion of C2 and C3 vertebrae with the presence of a rudimentary disc in between. C5 and C6 vertebrae showed partial fusion. D1, D2 and D7 level showed the presence of hemi vertebrae, while D5 and D6 showed butterfly vertebrae. The cervicodorsal region had marked kyphoscoliosis. The patient underwent posterior scoliosis correction with posterior instrumented fusion from D1 to D11 for the correction of double thoracic congenital scoliosis with D2 and D7 wedge vertebra (Figure 3). The patient was advised left pinna construction by plastic surgery for the cosmetic purpose to augment symmetry of face although it would not improve hearing ability of left ear. Furthermore, recent cardiac catheterization study showed severe obstruction across pulmonary homograft with grade II Pulmonary Regurgitation and hypertensive dysfunctional right ventricle warranting yet another surgery for homograft replacement. The patient s parents gave informed consent for reporting herradiography and clinical. | goldenhar syndrome, cardiac syndromes, case report, facio-auriculo-vertebral dysplasia, female | Not supported with pagination yet | null |
PMC10390344_01 | Female | 52 | In July 2022, a 52-year-old woman was diagnosed with SLE, lupus nephritis, and polyserositis. She presented with symptoms of facial edema, anorexia, nausea, abdominal distention, cough, and white phlegm. The severity of her condition was measured using the SLE Disease Activity Index (SLEDAI), which scored 20. Initially, the patient received treatment with glucocorticoids (methylprednisolone, prednisone), immunosuppressants (cyclosporine, mycophenolate mofetil), and hydroxychloroquine sulfate. After confirming no contraindications such as infection or allergies, the patient began receiving belimumab (480mg) on July 22, 2022. Four rounds of therapy were completed by September 29, 2022. During this period, the patient's symptoms gradually improved, and her SLEDAI score decreased. As a result, the dosage of glucocorticoids and mycophenolate mofetil was gradually reduced. However, in October, the patient developed an infection with the herpes zoster virus. Although she did not experience a noticeable cough or expectoration, she had occasional chest tightness and mild shortness of breath. Before the fifth belimumab therapy, on November 1, 2022, a chest computed tomography scan revealed patchy, nodular, and flocculent ground-glass density shadows in all lung lobes, as well as nodules and cavities in the dorsal segment of the left lower lobe. These findings indicated pulmonary infiltration associated with SLE and pulmonary infectious lesions (Figure 1a and b). HIV and anti-IFN-gamma autoantibody test results were negative. White blood count and C-reactive protein were normal. Serum beta-D-glucan, serum galactomannan, cryptococcal capsular antigen, tuberculosis antibody, candida IgG antibody and candida antigen test were negative. CD4+T lymphocyte count was 332/ul (reference range, 180-324/ul), CD8+T lymphocyte count was 646/ul (reference range, 1185-1901/ul), absolute NK cell count was 16/ul (reference range, 200-587/ul), IgG 5.44 g/L (reference range, 8-18g/L), IgA 1.16g/L (reference range, 0.9-4.5g/L), IgM 0.64g/L (reference range, 0.84-1.32g/L). The next generation sequencing (NGS) of bronchoalveolar lavage fluid (BALF) showed that Pneumocystis jirovecii (sequence number 178940) and Talaromyces marneffei (sequence number 7480). TM was cultured from the patients' BALF (Figure 2a and b). Meanwhile, BALF was positive for PJ by polymerase chain reaction (PCR). Finally, we discontinued the fifth belimumab therapy. Voriconazole (0.6g/d on day 1 and 0.4g/d from day 2) was used to treat TM infection. She had a history of glucose-6-phosphate dehydrogenase deficiency. Therefore, PJ pneumonia was treated with clarithromycin 1.8g/d for the first week. Our test for glucose-6-phosphate dehydrogenase was normal. Clarithromycin was changed to sulfamethoxazole. The treatment of SLE, maintenance of prednisone 5mg/d, MMF 0.75g/d, hydroxychloroquine sulfate 0.2g/d. After about one month of therapy, the reexamination of chest computed tomography showed that the pneumonia was improved (Figure 1c and d). The patient is still under follow-up. | pneumocystis jirovecii, talaromyces marneffei, belimumab, co-infection, systemic lupus erythematosus | Not supported with pagination yet | null |
PMC4467051_03 | Female | 0 | Maximize treatment success among Syrian refugees for those remaining in Jordan or moving to other countries. Four activities were undertaken: i) DOTS was instituted using Jordan NTP case management forms; ii) contingency plans for movement to other countries for treatment were developed; however, refugees were counseled to complete treatment in Jordan and to sign a modified World Care Counsel patient charter found in the CDC's Evaluation Tool for Tuberculosis Programs in Resource-limited, Refugee and Post-Conflict Settings, version 2. For consenting patients, WHO and IOM agreed to assist with transfers. At time of treatment initiation, the refugee was given a DOTS card updated with regimen administered, doses given, and pertinent laboratory results, which s/he could take to other treatment locations, either within Jordan or other countries; iii) psychosocial support was provided by the Jordanian Anti TB Association (JATA) to increase treatment success, alleviate fears, and reduce stigma. TB patients often deal with stigma; in addition, refugees have often witnessed traumatic events and struggle with the consequences of displacement adding to their need for psychosocial support; and iv) ongoing monitoring and evaluation. IOM collected TB cohort data and Jordan NTP assessed their governorate and national diagnostic TB centers using CDC's TB program evaluation tool for resource-limited and refugee settings. For the cohort analysis, the objectives were to verify that at least 85 % of the new smear-positive TB cases were cured.
Increase TB awareness and knowledge of treatment services among Syrian refugees and their healthcare providers by information, education and communication (IEC) material for camp and non-camp settings, and
Support the development and implementation of national guidelines for management of latent TB infection.
The strategic objectives were:
Each of the objectives had activities with indicators and responsible partner(s) for collecting those indicators. These indicators were collected by IOM and NTP and shared with UNHCR and WHO.
The strategy was implemented in July 2013 (Fig. 1). Of the five strategic objectives, objective 4: increase TB awareness and knowledge of treatment services was to ensure that the Syrian refugees with suspected TB could enter the Jordan TB control program. This objective had activities for refugees and for healthcare providers. IEC material were developed for both groups to increase awareness of TB signs and symptoms with messaging for refugees that TB is curable and where to seek care if someone had symptoms suggestive of TB. IOM conducted hour-long TB awareness sessions in the camps and community with the aid of a local medical association and collected the numbers of refugees attending by age group and sex. This strategy for the first 6 months of 2014, resulted in almost 61,000 (10 % of total) Syrian refugees attending IOM conducted TB awareness sessions, with equal proportions of males and females (Table 1). As far as anecdotal accounts, several refugees presented to IOM reporting that they had TB signs and symptoms after reading the IEC materials or attending the awareness raising sessions, while others when asked did not report having any sign and symptoms presumably because of fear of stigma within their community with a smaller fear of being deported (exact numbers not captured).
Second in importance was objective 1: increase TB screening, which offered Syrian refugees screening for TB symptoms, and if present, chest radiography and microscopic examinations, if indicated. With assistance of Jordan NTP and with UNHCR for newly arriving refugees, IOM screened almost 69,000 (11 % of total population) in first 6 months of 2014 (Table 1). Of those Syrians screened through June 2014, 45 % were 14 years of age or younger, which was comparable to the 45 % of the Syrian refugees aged 0 through 14 years derived from UNHCR regional demographic age structure. Therefore, this suggests children were captured in the TB screening efforts.
For objective 3: maximize treatment success, IOM collected, maintained, and reported TB data among Syrian refugees. Examining the 2013-2014 TB cohort data for Syrian refugees on treatment, for the first three quarters (January-March 2013, April-June 2013, July-September 2013 followed for 9 months each), 91 %, 96 %, and 85 % had completed their anti-TB treatment (or been cured) for an overall completion rate of 91 % (Table 2), with one death and no failures. For the first quarter, there was a 9 % (two persons) treatment default rate; however, zero for the following two cohort periods. These initial two persons returned to Syria and were lost to follow-up. Subsequently, IOM increased its assistance to the refugees under treatment and emphasized with them the importance of treatment completion before travelling.
For objective 2: increase TB diagnosis, for the first 6 months of 2014, of the 33 Syrian refugees with culture confirmed active TB disease, one each was diagnosed in Syria and Kuwait arriving with treatment and three were diagnosed in Syria coming without any treatment; all five once in Jordan received testing and appropriate treatment begun or was continued. Only three were found to be highly infectious with sputum smears containing Mycobacterium tuberculosis bacilli identified microscopically (smear-positive), 18 (78.3 %) were sputum smear-negative, and two (6 %) were children under 5 years of age (Table 3). The high percentage of smear-negative TB cases was because a) some patients were partially treated in Syria before coming to Jordan but were considered new cases once in Jordan, and b) with the highly dynamic movement of refugees within Jordan and the fear of losing cases, NTP increased the clinical diagnosis of TB to include smear-negative TB cases who had three additional criteria: (i) cough for more than 3 weeks, (ii) no response to an non-TB antibiotics, and (iii) a chest radiograph compatible with TB.
The public health strategy since initial development has identified TB cases among Syrian refugees at a higher rate (1.01 per 100,000 monthly or 12.17 per 100,000 for July 2013 through June 2014), than the pre-strategy period (0.73 per 100,000 monthly or 8.75 per 100,000 for 12 months), or approximately 40 % greater, using UNHCR population estimates (Table 4); more than double the Jordan TB incidence (5.8 per 100,000) for 2012. | jordan, refugees, syrian crisis, tb control program, tuberculosis | Not supported with pagination yet | null |
PMC6015715_01 | Male | 33 | A 33-year-old man was involved in an automobile accident and was brought to our hospital by ambulance. He had been in the front passenger seat and had been wearing a three-point seatbelt. He reported severe back pain, but showed no neurological deficit.
Anteroposterior and lateral radiographs of the spine showed an increased gap between the 1st and 2nd lumbar spinous processes and 2nd lumbar vertebral fracture (figures not shown). Magnetic resonance imaging (MRI) of the spine also demonstrated an L2 vertebral fracture and disruptions of the posterior ligamentous complex between L1 and L2, in combination with extensive subcutaneous hematoma (Figure 1).
Computed tomography (CT) of the spine in the sagittal orientation and 3-dimensional (3D) CT further revealed the involvement of the superior end plate of the L2 vertebra, comprising horizontal splitting from the left pedicle, through the left transverse process, and reaching the center of the neural arch (Figure 2). The right-sided L2 pedicle was intact.
After checking the general condition of the patient and excluding intra-abdominal injury by enhanced CT and ultrasonography, the patient underwent L1-L2 single-level instrumented fusion using a posterior approach. Initially, monoaxial pedicle screws with conventional trajectory were placed at L1 and L2 pedicles on the right side (intact pedicle side). A rod slightly bent in lordosis was then introduced and connected with these pedicle screws with a compression force applied between screws. This procedure achieved reduction and the fracture gap at the left L2 pedicle and lamina was completely closed. Polyaxial pedicle screws were used on the left side. A pedicle screw with a conventional trajectory was placed at the left L1 pedicle. A CBT pedicle screw was then inserted through the fractured L2 pedicle under fluoroscopy. This CBT screw was used as an alternative to an osteosynthesis screw. A rod was introduced on the left side, bilateral facet fusion with local bones obtained from the lower one-third of the L1 spinous process was performed, and the wound was closed. Although the merits of cross connectors for CBT screws remain unclear, we applied the connector in this case because connecting bilateral pedicle screws along conventional trajectories has been reported to increase the pullout strength of these screws. Postoperative X-rays showed good reduction by this single-level fixation (Figure 3).
The postoperative period was uneventful. Although rigid fixation was obtained with this procedure, a thoracolumbosacral orthosis (TLSO) was applied for 6 weeks, since this case was our first experience. Physical activities were not restricted with the TLSO. Sagittal CT and 3D-CT obtained at 6 months and 1 year postoperatively showed proper trajectory of the CBT pedicle screw and complete bone union (Figure 4). | null | Not supported with pagination yet | null |
PMC7332998_01 | Male | 45 | A 45-year-old man with no relevant past medical history presented to the eye casualty service complaining of sudden onset central 'white ring' and decreased vision in the right eye (RE) over the past seven days. Best-corrected visual acuity (BCVA) was 6/12 in the right eye and 6/6 in the left eye (LE). Intraocular pressure was 14 mmHg in both eyes. Examination of the RE showed no cells in the right anterior chamber and 1+ vitreous cells; fundus examination revealed a yellow placoid lesion involving the macular area with no signs of vasculitis or retinal necrosis. Examination of the LE was unremarkable.
MMI of the retina including colour fundus photograph, fundus autofluorescence, SD-OCT, fluorescein angiography and indocyanine green angiography are presented in Figure 1 (Fig. 1), Figure 2 (Fig. 2), and Figure 3 (Fig. 3).
The medical history was carefully reviewed; the patient admitted to be addicted to poppers and cocaine, and reported promiscuous homosexual activity over the last months. He denied intravenous drug use and any systemic symptoms such as headache, skin rash, nausea, weight loss, cough, or night sweats. A complete laboratory work-up was ordered, including TB QuantiFERON-TB testing, syphilis serology and human immunodeficiency virus (HIV) antibodies. Seven days after presentation, the patient reported spontaneous improvement in the vision of the RE, and BCVA improved to 6/9 in the RE and was stable in the LE. Full blood count, liver function test, kidney function, angiotensin-converting enzyme level were within normal range, HIV antibodies were negative. However, results for QuantiFERON-TB testing and syphilis had not been available yet. MMI revealed spontaneous improvement of the placoid lesion (Figure 2 (Fig. 2), Figure 3 (Fig. 3)).
Two weeks after presentation, BCVA further improved to 6/6 in the RE and MMI showed signs of early resolution of the placoid lesion. Laboratory results returned negative for QuantiFERON-TB testing, and positive for venereal disease research laboratory test and fluorescent treponemal antibody testing. Therefore, a definite diagnosis of ASPPC was made, and the patient was promptly referred to the Infectious Disease Department for systemic treatment with penicillin. | acute syphilitic posterior placoid chorioretinitis, natural course, pathogenesis, retinal imaging, syphilis | Not supported with pagination yet | null |
PMC9234807_02 | Male | 43 | A 43-year-old man was admitted at the emergency room of the Sardjito General Hospital with a chief complaint of rash on the overall body. The ethnicity was Javanese, based on the ethnicity of parents and both grandparents. Vital sign examination showed an elevated blood pressure of 170/100 mmHg, heart rate of 80 beats/min, respiratory rate of 20/min and normal temperature.
Three days prior to the admission, he complained of fever, headache, muscle pain, and took over-the-counter medicine. Two days before the admission, rashes occurred on the face in addition to red eyes and mouth ulcers. These symptoms got worse on the day before the admission when the rash spread to the overall body. Physical examination showed erythema on the overall body (more than 30%), eye discharge, and mouth ulcers (Figure 1). Laboratory examination showed an increased leukocyte count with increased percentage of neutrophil, decreased erythrocyte counts, and markedly elevated liver enzymes and renal function tests (Table 1).
The diagnosis of SJS was established based on the skin and mucosal involvement and positive Nicolsky's sign. One month before the admission, the patient had started allopurinol and antihypertensive medication. The patient had a history of hypertension in the last year before the complaints started. The patient was administered methylprednisolone of 31.25 mg/24 h. Allopurinol was discontinued, but antihypertensive medications were continued. Dressing with NaCl 0.9% was given for 15 min twice a day. At discharge, the patient did not show either new erythematous patches or vesiculobullous lesions.
Three ml peripheral blood was sampled from both patients by using an EDTA-containing vacutainer from each patient. Blood samples were transported to the Molecular Biology Laboratory of the Faculty of Medicine, Public Health and Nursing Universitas Gadjah Mada. Buffycoat was isolated from blood samples by 1500 RPM cold centrifugation for 10 min. Further, DNA was extracted from 100 ul of buffycoat by using a QIAamp(R) DNA minikit (Qiagen, Switzerland) by following the instruction manual. The quality of extracted DNA was analyzed through DNA electrophoresis with the objective to observe single genomic DNA as well as 260/280 nm absorbance ratio of >=1.75 by using a spectrophotometer (DeNovix DS-11). The quantity of DNA was obtained through multiplication of 50 ug/ml x 260 nm spectrophotometer read-out x dilution factor. Further, purified DNA was stored in a -20 C freezer until used. Purified DNA was transported in cold chain to the School of Medicine and Health Sciences, Atma Jaya Catholic University of Indonesia, for further downstream genomic analysis.
HLA-B*58:01 was determined by using the ExProbe HLA B*58:01 Typing Kit (TBG Biotechnology Corp., Taipei, Taiwan) following its instruction manual. The detection system was based on the use of a real-time quantitative polymerase chain reaction (qPCR) technique containing primer mixes and SYBR green dyes. The presence of amplification is detected by activation of fluorescence and a positive indication of the existence of allele specific sequence within the genomic DNA. An internal control primer pair that amplifies a conserved region of the housekeeping gene, cystic fibrosis gene, was included in the PCR reaction mix, and the PCR product of the internal control primer pair serves as an indication of the integrity of the PCR reaction. A negative control was also included in the kit, which should provide a negative result after the completion of the PCR.
For each PCR reaction, a mix of 4.5 ul primer mix, 10.5 ul qPCR buffer mix, and 3 ul of template DNA was made, containing either positive or negative controls or purified DNA at concentrations ranging from 5 to 40 ng/mul. The thermal cycling was run using the BioRad CFX96 Real Time Detection System programmed as follows: one cycle of heating at 95 C for 3 min, 36 cycles of denaturation at 93 C for 30 s, annealing at 62 C for 45 s, and elongation at 72 C for 40 s. An additional step done for data collection included recording Melt Curve between 65 C and 95 C for 15 s, at increments of 0.5 C. Upon run completion, the melt peak threshold was set at 40 -dRFU/dT to determine melt temperature when peaks were present.
Indication of the HLA-B*58:01 allele was observed as the presence of peaks in the melt curve plot profile of the internal control and target allele region at 73.5 C-79 C and 88.5 C-90 C, respectively, or a single peak at 88.5 C-90 C. Whereas the absence of HLA-B*58:01 is shown by a presence of peak at 73.5 C-79 C denoting internal control only, but not the target allele region. HLA-B*58:01 typing of both samples showed positive results (Figures 2, 3).
Ethical approval was obtained from the Medical Research Ethics Committee at Universitas Gadjah Mada no KE/FK/1150/EC. Written consent was obtained from each patient after receiving adequate information about the study. | hla-b*58 | Not supported with pagination yet | null |
PMC10013466_01 | Male | 27 | A 27-year-old male sought the dermatology care service for a skin lesion located on the right mandible, characterized by increased volume, erythema, and fistulous tracts. The dermatosis comprised an area of 3x4 cm and exuded a slightly seropurulent secretion (Figure 1A). He claimed it had been evolving for more than five months and mentioned having undergone previous treatment with different topical and oral antibiotics, without improvement. He claimed to be a permanent resident in Mexico City (19 25'10"N 99 08'44"O), but he had made frequent business trips to Veracruz state (19 26'05"N 96 22'59"O) in the last year for periods of one to two weeks. The suspected clinical diagnoses were actinomycosis, cutaneous tuberculosis, or sporotrichosis. Thus, a skin biopsy, histopathological analysis, and culture from a tissue sample were performed. Also, a blood count and blood chemistry were requested.
Regarding histopathology, the hematoxylin and eosin stain exhibited an epidermal hyperplasia and ulceration. Tissue slices (Figure 1B) exhibited an inflammatory infiltrate composed of histiocytes, plasma cells, lymphocytes, and polymorphonuclear located in the dermis and subcutaneous tissue (Figure 1C). There was also vascular damage. No fungal structure or mycobacteria was observed in special stains such as Gram, Ziehl-Neelsen, PAS [periodic acid-Schiff stain], and Giemsa. No fungal growth was observed in the culture media (Sabouraud Dextrose Agar with Cycloheximide and Chloramphenicol [Mycobiotic Agar], potato dextrose agar, and brain heart infusion). Surprisingly, parasites (amastigotes and trypomastigotes), as well as intracellular bacteria, were presented in the tissue samples (Figures 1D, 1E, and 1F). Furthermore, the bacterial culture analyses showed evidence for the growth of two different isolates. The former corresponded to a Gram-negative bacillus, Ziehl-Neelsen negative, and catalase-negative, identified by PCR and sequencing as Bacillus velenzensis (Figure 1G), and the second was identified as Corynebacterium sp. The antibiogram reported a Bacillus sp., sensitive to cephalothin, ciprofloxacin, fosfomycin, gentamicin, tetracycline and oxycline, resistant to nitrofurantoin and trimethoprim/sulfamethoxazole, and intermedium resistant to clindamycin, erythromycin, and penicillin. Moreover, culture media for fungi were evaluated weekly and did not present any growth during five weeks. Consequently, serological diagnoses were requested to confirm the diagnosis of Chagas disease or Leishmania sp. The initial tests, executed in 3 independent laboratories (blood bank laboratory [chemiluminescence architect Chagas kit (Abbott)], a research laboratory specialized in parasitic infections in our institution [ELISA home with the Ninoa strain as antigen] , as well as an external private laboratory [chemiluminescence technique]), were reported as negative.
PCR analysis was done to identify Leishmania sp. and specific Leishmania species with DNA isolated from the tissue biopsies and blood; these tests showed negative results. However, a PCR established with the DNA satellite showed the presence of Trypanosoma cruzi DNA . The sample sequence showed an identity of 99% with the T. cruzi ClBE07 clone (GenBank No AY520036). Furthermore, Bayesian phylogenetic analysis (Figure 2) supported the grouping of the sequence obtained inside T. cruzi DTUs and showed high posterior probability values (1.00) for DTU II. On the other hand, PCR-sequencing analysis to identify bacterial 16S revealed the presence of Bacillus amyloliquefaciens, also known as velezensis, in the tissue samples and culture (GenBank ascension No ON737906).
Laboratory tests were requested, including blood count and chemistry, reporting neutrophilia and eosinophilia. Other parameters were within the average reference values. Also, a complete cardiac assessment was performed, including thorax radiography, an electrocardiogram, and an echocardiogram. All tests were reported within the standard values. In addition, two independent laboratories (public and private) repeated ELISA and Western blot during the patient follow-up, and the results were positive four months after the original assessment.
The initial reports established a mixed cutaneous infection; thus, according to the antibiogram report, the patient received standard doses of cefalexin and ciprofloxacin with clinical improvement in three weeks. However, according to PCR and histology results that established Chagas disease (CD), the patient was referred to the Tropical Medicine Center, an authorized center for the diagnosis and management of Chagas disease in Mexico , because the antiparasitic treatment for CD is not commercially available in Mexico. In addition, the CD is a mandatory report disease, because only special health authorities are able to provide the treatment (Secretaria de Salud). Sadly, current Mexican health care system lineages for Chagas treatment do not allow established treatment if the serology is negative, so for our patient, the treatment was delayed for several months, despite the parasite being visualized in tissue slices and the infection being confirmed with a PCR test . Thus, the opportunity to treat the patient in the acute phase, when the benefit of treatment is more significant, was lost. Four months after the diagnosis, when the serology reported a positive result, treatment with nifurtimox was started, and doses of 240 mg at 8:00, and 180 mg at 15:00 and 22:00, were administered for 60 days. Afterwards, serology tests performed in August 2021 and January 2022 reported negative results. The last follow-up performed in July 2022 included a PCR and ELISA, which were also negative. | null | Not supported with pagination yet | null |
PMC5787533_01 | Female | 16 | A 16-year-old female from a Chinese family with consanguineous parents, developed her first episode of jerks after a cold 11 years ago. The jerks involved right foot and leg without preceding sensation and was not relieved until she was given diazepam intravenously. The antiepileptic drugs (AEDs) were provided, but the episodes still occurred 3-4 times/year. The jerks were usually triggered by fatigue, emotions, or fever and lasted for minutes to hours. It could only be controlled by intravenous diazepam. She was diagnosed with "psychogenic attack" 6 years ago and was weaned off AEDs. The "psychogenic attacks" continued. Three years ago, the patient experienced loss of consciousness for minutes after right arm jerks. Six days before the admission, she developed right foot jerks after a long walk, but the jerks could not be relieved completely by diazepam. The jerks stopped with sleep without any treatment and reoccurred with waking. During the course of the disease, the patient had no intellectual disability or developmental delay, hearing loss, onychodystrophy, osteodystrophy, paresthesia, or cerebellar ataxia. She felt fatigue after running, long-distance walking, and climbing stairs. She had no remarkable medical history. Another individual in her family had similar paroxysmal episodes of leg jerks. Upon physical examination, no neurological deficit was found. MMSE test scored 29. The audiometry test was normal. The serum autoimmune panel, including anti-streptolysin "O," C-reactive protein, rheumatoid factors, immunoglobulin G, immunoglobulin M, immunoglobulin A, alexin C3, alexin C4, erythrocyte sedimentation rate, anticardiolipin antibody, anti-nuclear antibody, anti-neutrophil cytoplasmic antibody, thyroid peroxidase antibody, and thyroglobulin antibody, was negative. Both amino acid and organic acid metabolism tests of serum and urine were negative.
Continuous video electroencephalogram (EEG) monitoring showed normal background without EEG change during leg jerking. One event was captured. The patient's head and eyes deviated to the right with the right arm rising and the leg stretching straight. Then, the convulsion involved all limbs and lasted 20 s. During the event, she was alert but could not speak. The EEG showed rhythmic activity from the left frontocentral and midline regions. The activity quickly spread to the contralateral side with increased amplitude (Figure 1A). The brain MRI was reported normal. The somatosensory-evoked potential (SEP) showed remarkable reduced amplitude in the left cortex when right leg was tested. The upper limbs' SEPs were normal. Genetic analysis through an epilepsy panel of 480 genes by Kangso Medical Inspection reported a homozygous mutation in the TBC1D24 gene, c.229_240del (p.82_84del). The homozygous mutation was inherited from her healthy parents who were heterozygous, which was consistent with autosomal recessive inheritance (Figure 1B and Supplementary Figure S1). We could not predict protein function related to this mutation through SIFT, Polyphen2, or Mutation Taster analysis software.
Morphometric analysis program was performed on 3-T T1-weighted images using SPM12 toolbox in MATLAB R2011b following previously established methods. The computed output was a junction map, which highlighted abnormal structures deviating from the average normal structures based on z-score. The normal database was provided along with the MAP program. The highlighted region might correlate with gray-white blurring on the underlying MRI. The MAP analysis identified a subtle abnormality candidate with a z-score over 4, a subtle gray-white blurring in the left medial parietal region (Figure 2).
The patient was given levetiracetam with a gradually increasing dosage to 1.25 g twice/day and sodium valproate with a dosage of 0.5 g twice/day and was completely relieved several days later. Upon follow-up, the patient was weaned off the sodium valproate and did not report any symptoms in the 10-month follow-up.
Both patient and parents provided written informed consent for the study and publication. | mri post-processing technique, tbc1d24 gene, epilepsia partialis continua, homozygous mutation, morphometric analysis program | Not supported with pagination yet | null |
PMC10331237_01 | Female | 31 | A 31-year-old female patient was admitted to our emergency room (ER) with a generalized tonic-clonic seizure. A fever, meaningless movements, restlessness, and self-talk complaints had started a week ago in her medical history. She had a history of surgery for ovarian teratoma 10 years ago. There was no history of drug use. Two polymerase chain reaction (PCR) tests for novel coronavirus disease-2019 performed within the last week were negative. During the initial neurological examination in our ER, she was in the postictal period; her general condition was moderate, her consciousness was drowsy, and cooperation was limited. There was no neck stiffness, pupils equally round, and reactive to light. There was not any facial asymmetry. Examination of other cranial nerves was normal. Spontaneous motor movements were observed in all extremities. Plantar reflexes were flexor bilaterally. Vital findings were stable.
Electrocardiography was regular with normal sinus rhythm. There was not any gross abnormality in hemogram and serum biochemistry tests. Lactate level was slightly elevated to 1.74 mmol/L (0.5-1.6 mmol/L). Brain computerized tomography (CT), magnetic resonance imaging (MRI), and MRI-venography were normal (Fig. 1).
There was not any sign of pneumonia on thorax CT. Due to recurrent seizures despite intravenous diazepam infusions in our ER, phenytoin infusion was introduced. A partial reduction in frequency and duration of the seizures were obtained after phenytoin infusion. Then, she was hospitalized in our intensive care unit. Intravenous phenytoin was maintained with the dosage of 100 mg t.i.d.
Electroencephalogram (EEG) revealed a generalized slow background activity with low voltage that was observed throughout the whole trace without any accompanying epileptiform discharge. Delta waves were observed in the left frontotemporal and central areas (Fig. 2).
During her follow-up, dexmedetomidine infusion was started to decrease her agitation. Opening pressure (17 cm H2O) was normal during the lumbar puncture. Leukocyte count was 10/mm3, erythrocyte count was 10/mm3, and protein level was 29 mg/dl in cerebrospinal fluid (CSF). Prophylactic ceftriaxone (2x2 g), vancomycin (2x1 g), and acyclovir (3x750 mg) were started upon the recommendations of our infectious disease department. Haloperidol and chlorpromazine treatments were initiated after psychiatry consultation requested for the consideration of her persistent agitation. Lorazepam was added during the follow-up. Extensive panel tests for meningitis were negative. CSF-PCR for tuberculosis, Gram's stain of sputum, acid-fast bacilli scan, venereal disease research laboratory-rapid, plasma reagin, treponema pallidum hemagglutination, Rose Bengal test for Brucella results were all negative. Serum ammonia level (77.7 micromol/L) was normal. Upon the persistence of her agitation, mirtazapine and quetiapine were added.
Autoimmune encephalitis panel assessed with immunofluorescence assay revealed NMDAR receptor antibodies in CSF. Intravenous immunoglobulins (IVIG) were administered as 25 g per day. After IVIG therapy for 5 days, her seizures were ceased. She was conscious and partially cooperative at the end of the 2nd week and was discharged. During the outpatient clinic follow-up at the end of 6 weeks, the patient did not report any recurrent seizures, and her neurological examination was completely normal. Rituximab treatment was introduced for long-term immunomodulation. | anti-n-methyl d-aspartate receptor encephalitis, norse, anti-n-methyl d-aspartate receptor encephalitis, cerebrospinal fluid, electroencephalogram, new-onset refractory status epilepticus | Not supported with pagination yet | null |
PMC8649689_01 | Unknown | 60 | The patient was a 60-year-old worker with repeated cough and dyspnea for ~10 years. The patient had a smoking history of 20 pack-years for 30 years and had chronic obstructive pulmonary disease and pulmonary bullae. Usually, these symptoms can be controlled using bronchodilators and inhaled corticosteroids; however, the dyspnea, cough, and sputum expectoration of the patient gradually worsened. The patient was admitted to our hospital for the first time due to sudden shortness of breath 1.5 years ago. Physical examination showed that the patient was thin, had clubbing digits, a slightly collapsed bridge of the nose, and had stunted auricles (Figures 1A-C). The chest was in a typical barrel shape, the intercostal space was widened, the breath sounds were lower, and wet rales can be heard in both lower lungs. Chest computed tomography (CT) showed reduced tracheal lumen, thickened tracheal wall, emphysema, and bilateral lung infections (Figures 2A-C). Pulmonary function tests revealed severe mixed ventilatory dysfunction, which was mainly obstructive, with a slight decrease in diffusion function (Figure 2H). Blood gas analysis suggested type 2 respiratory failure. Routine blood tests showed that white blood cells, neutrophils, and C-reactive protein were significantly elevated, suggesting an infection in the lungs. Although low-flow oxygen (2 L/min), antibiotics, glucocorticoids, and bronchodilators were administered, the patient's symptoms were not significantly relieved. A chest CT scan including the inspiratory and expiratory phases showed that the lumen of the trachea and main bronchus severely collapsed at the end of expiration compared with inspiration (Figures 2D-G). Subsequently, bronchoscopy showed that the mucosa of the trachea and main bronchi were severely hyperemic and swollen, and the cartilage had disappeared (Figures 3A-G). Although the lumen was normal during inhalation, the tracheal membrane protruded into the lumen during exhalation, resulting in the complete collapse of the lumen, and inability to eliminate secretions; these changes were not seen in the distal airway. A diagnosis of RP with TBM was highly suspected. Subsequently, a biopsy of the patient's auricular cartilage was performed. Pathology reports showed multiple necrotic chondrocytes accompanied by inflammatory cell infiltration (Figure 1D). Finally, the diagnosis was clear and consistent with our hypothesis. Subsequently, three nickel-titanium alloy coated memory stents were implanted in the trachea and bilateral main bronchus under local anesthesia (Figures 2I-K). Pulmonary function tests revealed moderate mixed ventilatory dysfunction, which was mainly obstructive, with a slight decrease in diffusion function (Figure 2L). Prednisone (1 mg/kg) was continued, and the patient was discharged. The patient was readmitted to our hospital for worsening dyspnea 1 year ago. Chest CT showed an unobstructed trachea and bilateral main bronchus, the stent was well-fixed, and the subglottis and upper part of the stent were slightly narrowed (Figures 2M-Q). Bronchoscopy showed that the lumen of the subglottis to the upper segment of the tracheal stent was narrow, the mucosa was severely swollen, cartilage had disappeared, and granulation hyperplasia was present (Figures 3H-L). The narrow lesion was significantly improved after bronchoscopy-guided argon plasma coagulation, and CO2 cryoablation was performed, which significantly relieved the patient's symptoms (Figures 3M-P). The patient was administered prednisone (1 mg/kg). Six months ago, the patient was readmitted to our hospital because of sudden dyspnea. Emergency chest CT and bronchoscopy showed granulation hyperplasia and scar tissue in the lumen of the subglottis to the upper segment of the tracheal stent, hyperemia and swelling of the mucosa, and a large amount of thick sputum blockage in the lumen, resulting in severe narrowing of the lumen (Figures 2R, 3Q). To avoid the risk of major airway bleeding and asphyxia during bronchoscopy under conventional ventilation, we decided to perform bronchoscopy-guided interventional therapy with VV-ECMO using a heparin-coated membrane lung.
We first percutaneously inserted a 22-Fr cannula into the left femoral vein and a 16-Fr venous cannula into the right internal jugular vein of the patient. The direction of the pipe connection was as follows: left femoral vein centrifugal pump membrane lung right internal jugular vein. The circulatory system was pre-filled with Wanwen 1,500 mL and continuously infused with heparin during ECMO. The mean arterial pressure, SpO2, hematocrit, and activated clotting time (ACT) during transfusion were monitored. The ECMO speed was 3,500 rpm, the blood flow velocity was 3 L/min, the average arterial pressure was maintained at 90 +- 10 mmHg, and ACT was maintained at 250 s. We performed bronchoscopy interventional therapy under general anesthesia with oxygen supply guaranteed by ECMO. For the intervention, we first used a CO2 cryotherapy instrument to remove the local granulation tissue during the operation and then a needle-shaped high-frequency electrosurgical knife to make a radial cut on the narrow opening (Figure 3R). Subsequently, balloon dilations were performed three times at the lesion. Finally, CO2 cryoablation was performed (Figure 3S). The total treatment time was 1 h, the intraoperative bleeding volume was ~50 mL, and the SPO2 was maintained at 90-95%; the rest of the vital signs were stable. After the operation, the patient's tracheal stenosis significantly improved, and the bronchoscope was able to enter the distal airway smoothly, enabling the aspiration of large amounts of viscous secretions; the airway stent was in a good position and no serious complications, such as rupture were observed (Figures 3T-X, 2S,T). After the operation, the patient was transferred to the ICU for monitoring. After 12 h, ECMO support was stopped, and the patient was implanted with a laryngeal mask and switched to a mechanical ventilator for oxygenation. On the second day after surgery, we removed the laryngeal mask and switched to non-invasive ventilator-assisted ventilation. On the fourth day, the patient was discharged from the hospital. However, the patient eventually died due to sudden respiratory failure during half year follow-up. Clinical history of the patient can be seen in Figure 4. | bronchoscopy, extracorporeal membrane oxygenation, interventional therapy, relapsing polychondritis, severe airway stenosis, tracheobronchomalacia | Not supported with pagination yet | null |
PMC9175977_01 | Male | 69 | A 69-year-old male ex-smoker (five packs/year) presented to our clinic with a routine spirometry showing a mild restriction (FEV1=2.11 l, 72 % of predicted; FVC=2.69 l, 69 % of predicted; FEV1/FVC=0.78). He had no respiratory complaints, including cough, sputum production or dyspnoea, and denied any other symptoms, such as malaise, fever or weight loss. His medical history included hypertension, diabetes and dyslipidaemia, for which he received medications, and past surgeries for nephrolithiasis, ocular melanoma and basal cell carcinoma of the scalp. Ten years prior to presentation he had a positive tuberculin skin test (17 mm), for which he was not treated.
Physical examination revealed diminished breath sounds and dullness to percussion in the left lung base, and the chest roentgenogram showed a medium-sized left pleural effusion. Laboratory tests showed a C-reactive protein (CRP) level of 53 mg dl-1 (reference range, 0 to 5). Complete blood count, including leukocyte and differential count, was normal (9460 cell mul-1 total leukocytes with 59 % neutrophils, 30 % lymphocytes, 9 % monocytes,and 1 % each of eosinophils and basophils) and so were his renal and liver functions. An FDG-PET/CT was performed, which showed a left pleural effusion with FDG-avid thickening of the basal dorsal pleura (shown in Fig. 1). There neither lymphadenopathy nor other abnormalities in the lungs or in the rest of the body.
Thoracocentesis was performed and a total of 700 cc of clear fluid was drained. Analysis revealed an exudate with lymphocyte predominance (93 %). Lactic dehydrogenase (LDH) and protein levels were mildly elevated. Adenosine deaminase level was not measured. Cytological examination did not show malignant cells. No bacteria were detected in bacterial cultures or by Ziehl-Neelsen staining of the fluid and polymerase chain reaction (PCR) for TB was negative. Serum immunoglobulin and anti-interferon-gamma auto-antibody levels were not performed. However, M. abscessus was isolated in one out of three sputum cultures.
Thoracoscopic inspection of the left pleural cavity revealed adhesions at the lung base, in addition to multiple small white lesions of the parietal pleura in this area. Pleural biopsies were taken, which demonstrated well-formed epithelioid and giant cell granulomas with tiny foci of central necrosis in some of them, consistent with granulomatous pleuritis (shown in Fig. 2). Cultures that were taken from the pleura and from the effusion during the surgery grew M. xenopi . Consequently, the M. abscessus in the sputum was considered a contamination, and the patient was diagnosed with granulomatous pleuritis due to M. xenopi .
Antimycobacterial therapy was initiated with isoniazid, rifampin and ethambutol. However, treatment was stopped after 2 months due to arthralgias. By that time, the serum CRP level was 2 mg dl-1 and a repeat chest CT showed resorption of the pleural effusion with some residual pleural thickening. At the last follow-up 9 months after the initial presentation, the patient feels well and a chest radiograph shows mild pleural thickening. | mycobacterium xenopi, pleuritis, pulmonary infection | Not supported with pagination yet | null |
PMC8651025_01 | Male | 33 | Case 1 - A 33-year-old male patient presented at the refractive surgery clinic with complaints of decreased visual acuity in the right eye. Bilateral FS LASIK had been performed 12 years earlier. His uncorrected visual acuity (UCVA) was 20/200 in the right eye and 20/20 in the left eye. Manifest refraction values were -1.25 x65 in the right eye and -0.25 in the left eye. The BCVA in his right eye was 20/63. Corneal topography (Pentacam; Oculus Optikgerate GmbH, Wetzlar, Germany) findings were compatible with corneal ectasia in the right eye and a postmyopic ablation pattern in the left eye (Fig. 1). A Corvis ST and Pentacam biomechanical/tomographic assessment was performed. All of the topographic and biomechanical index values are listed in Table 1. Although the topographical and clinical findings were not consistent with corneal ectasia in left eye, the CBI, TBI, and BAD-D values suggested biomechanical weakness (Fig. 2). Corneal crosslinking (CXL) was planned for the right eye. It was not pursued immediately for left eye since the UCVA had not deteriorated. The patient was informed about the possibility of ectasia development in the left eye, follow-up was recommended, and he was advised not to rub his eyes. | biomechanics, corvis st, femtosecond-assisted laser in situ keratomileusis, postlasik ectasia | Not supported with pagination yet | null |
PMC9728865_01 | Female | 17 | On May 25, 2020, a 17-year-old girl named Darnella Frazier used her smartphone to film Minneapolis police officer Derek Chauvin kneeling on the neck of George Floyd for more than nine minutes. She uploaded the footage of the fatal incident to Facebook. Within two days, the video went viral, sparking global outcry. By June 2, 2020, Brianna Agyemang and Jamila Thomas, two Black women and music executives at Atlantic Records, created the viral hashtag #TheShowMustBePaused. They called on recording artists to use their platforms to draw attention to systemic racism by posting a single Black square on their Facebook and Instagram timelines. Millions of everyday people joined the initiative and did the same in what came to be known as #BlackOutTuesday.
This study provides a snapshot of the Instagram activity leading up to this viral moment, tracking it through its apex and eventual nadir. Our investigation is novel, insofar as #BlackOutTuesday likely marks the first time in the history of social media movements that a visual platform:not a predominantly text-based one like Twitter:took center stage. Twitter has been well-studied since the Arab Spring uprisings of 2011. Most of these investigations have focused on textual diffusion, however, ignoring the adage that a picture is worth a thousand words. In modern social movements, it is often an incendiary photo or video that galvanizes the public's support for a social justice issue. These images can seem fleeting, however, because ephemeral platforms such as Instagram feature videos that disappear after 24 hours and an endless scroll of content that can be hard to search. This study, therefore, answers a recent call for media scholars to document more visual social media content for scholarly analysis. The data collection we carried yielded a corpus of almost 1.7 million photos obtained during the June 2020 Black Lives Matter protests.
Our review of prior literature features three parts. First, we explain how (and why) Twitter was invaluable to the first wave of the Black Lives Matter movement, while its second wave shifted toward Instagram, according to extensive qualitative work. We then summarize the evolution of Instagram studies to emphasize further why we chose to highlight this emergent site for protest journalism. Lastly, we consider the press concerns of protests and the broader theory of connective action, namely how modality and affordances impact frames of legitimacy and the emergent opinion leaders. Through this frame, our results demonstrate the precarious nature of movement media, and how content creators, journalists, and everyday Instagram users co-evolved their earlier social media practices to report on and shape one of America's largest-ever human rights movements.
The Black Lives Matter movement began in 2013, when George Zimmerman was acquitted for shooting and killing Trayvon Martin, an unarmed Black teenager, in Florida. Alicia Garza, a well-known political organizer from Oakland, California, wrote a love letter to Black people after the verdict was announced and posted it on Facebook. She ended it with the sentence, "Black Lives Matter." Her friend and fellow organizer, Patrisse Cullors, added a hashtag to the front of the message and re-broadcast it on Twitter. It would be one more year before the #BlackLivesMatter hashtag went viral in 2014. On August 9, a White police officer, Darren Wilson, shot and killed an unarmed Black teenager, Mike Brown, in Ferguson, Missouri. When police allowed Brown's body to lay in the road for four hours, uncovered in the sweltering summer heat, peaceful protests began. Citizen journalists used the hashtag to document Brown's killing and the community's outrage. Activists used the #BlackLivesMatter hashtag to call on the police department to cover up Brown out of respect for his humanity. For the rest of the summer, use of the hashtag soared.
Groups that opposed the new Black Lives Matter movement created counternarrative hashtags, such as #AllLivesMatter (to reject claims of racism in policing) and #BlueLivesMatter (to support law enforcement). An exhaustive study of Twitter discourse found that six major communities relied on Twitter to discuss police brutality in 2014 and 2015: Black Lives Matter activists, Black entertainers, conservatives, bipartisan reporters, legacy media outlets, and young Black Twitter users. Freelon, McIlwain, and Clark (2016) analyzed 40.8 million tweets, more than 100,000 web links, and 40 interviews with frontline activists and allies, and discovered that the vast majority of those who tweeted using the #BlackLivesMatter hashtag denounced police brutality. The authors also found that activists who tweeted movement-related news succeeded in educating "casual observers" who either expressed "awe and disbelief at the violent police reactions to the Ferguson protests" or "conservative admissions of police brutality," especially in the cases of Eric Garner and Walter Scott's public police killings. Overall, the research postulated that activists' primary goals for using Twitter were "education, amplification of marginalized voices, and structural police reform".
Subsequent inquiries into the Black Lives Matter movement's use of Twitter in 2016 included how users craft counternarratives to anti-Black racism, how Black feminists hijacked the platform to highlight intersectional struggles, and even how Millennial organizers were departing from the Civil Rights Movement's protest templates to create their own. The Pew Center, for example, reported in 2016 that the #BlackLivesMatter hashtag peaked during the 10 days spanning July 7-17:with nearly 500,000 tweets of the back-to-back killings of Alton Sterlingaily. Studies such as these centered around Twitter as a digital public sphere. In the book Hashtag Activism, for example, the authors argue that the Black Lives Matter movement, and other contemporary Twitter-based movements then, were propelled by strong Twitter communities of Black women. The #GirlLikeUs transgender justice campaign, or the #SayHerName movement to raise awareness about Black women victims of police brutality, flourished and thrived on the platform. As the decade came to a close, the Black Lives Matter movement waned. Although publications such as The New York Times or The Atlantic published end-of-decade pieces on how social media shaped modern protest in the 2010s, the emphasis remained on Twitter's impact:until this study.
In May 2020, Darnella Frazier's cellphone video of George Floyd's murder reinvigorated the Black Lives Matter movement:in the same way that the photographs of Emmett Till's 1955 lynching rebooted the dwindling, post-World War II Civil Rights Movement. In Bearing Witness While Black, Richardson draws these parallels, arguing that pictures and videos have played an outsized role in Black movement-building. Richardson also documents the rise in smartphone video as a tool for political testimony to explain why Black Americans "press record" as a means to fight back against systemic oppression in the US.
Media pundits and scholars have in recent years begun exploring Instagram's affordances, and early content largely fell outside of the scope of social movement media. An early probe into the world of Instagram endeavored to explain how users' "experiences of production, sharing, and interaction with the media they create" are mediated by the "interfaces of particular social media platforms". This study was one of the first to use computational analysis and visualizations to explore Instagram's social and cultural patterns. The team compared the visual signatures of 13 different global cities using 2.3 million Instagram photos, and honed in on 200,000 Instagram photos that were uploaded in Tel Aviv, Israel. While the three-month study confirmed that one could ascertain people's activities and political habits from the geotagged photos, there was not a sustained look at a particular viral moment.
Other Instagram studies from the 2010s followed a similar pattern. Scholars often investigated what images people uploaded to Instagram or how to detect the age of a user from their photos, but stopped short of analyzing Instagram photos during moments of political unrest. Still other studies elucidated the effects of pop culture, or the five primary social and psychological motives of Instagram use: "social interaction, archiving, self-expression, escapism, and peeking". Recent studies on political movements followed en suite. Meme pages and celebrities, for instance, have been recently characterized to spark instances of political participation in Morocco, maintain partisan identity in Canada, promote public health behavior, or facilitate misinformation. Recent work has focused on the growing role of visuals in protests directly, such as the case of the Hong Kong Protests in 2019. Amid the virality of the #BlackOutTuesday hashtag on Instagram we decided to study the visual platform, finally, in a political context.
The backdrop of the global discourse about Mr. Floyd's controversial video was not the only reason for the shift away from Twitter and toward Instagram. Instagram content is four times more likely to be geotagged than Twitter content, which provides us invaluable insight into when and where the #BlackOutTuesday groundswell occurred. Instagram provided the right lens for studying the visual dimension of this phenomenon. Recent studies have also shown the importance of racial presentation in mediating (mis)information dissemination, which may be more evident in the visual mode. Furthermore, the Twitter discourse we analyzed seemed more like "ambient journalism" that was always on, rather than the more intentional educational and call-to-action-style posts that we observed casually on Instagram.
Lastly, we shifted our focus toward Instagram because the people did too. Many of the #BlackOutTuesday posts on Twitter linked back to an original post on Instagram, using either Facebook's native algorithmic syncing tool or the IFTTT (IfThisThenThat) application. We were also intrigued by Instagram's many technological affordances. The platform remains a walled garden, which does not make it easy for users to hyperlink outward. That is, Twitter can reference Instagram posts but not vice versa, creating an asymmetry in information flow. Trapping captive audiences within Instagram gives the platform enormous power over what the user sees:in that content is mediated solely through one platform. Without trigger warnings or decency screens over controversial content, Instagram users in June 2020 faced an increased likelihood of viewing potentially harmful content while scrolling through their feeds. Likewise, users found that they were beholden also to Instagram's opaque user guidelines around protest posts. A USA Today piece reported that Black users were suspended from Facebook for even talking about racism. Similarly, Instagram and TikTok apologized for algorithmically silencing Black voices.
Our last preoccupation with Instagram during the #BlackOutTuseday campaign centered around protesters who were captured in the viral pictures. Their visibility meant that their chances of being added to police's facial recognition databases or other forms of AI-based cataloguing grew. Much like the 2019 Anti-Extradition Law protests in Hong Kong:when activists combatted doxxing by communicating through an high-level encryption app, Telegram:we observed users creating 24-hour ephemeral Instagram "Stories" that were designed to disappear, presumably to limit the retrieval of their movement media.
There is certainly the perspective that #BlackOutTuesday was performative and did not directly advance the movement. For instance, a recent study found, through 20 interviews of wellness influencers, that sharing of the square was for maintaining credibility with their following base. However, their work focuses primarily on influencers, users that have achieved a certain level of popularity, and in wellness, which is an even more specific slice of Instagram. Moreover, the greater point is that there was sufficient demand for influencers to navigate the movement, which creates a larger presence on Instagram than previously, whether solidarity is performative or not.
Modality, taken from semiotics, refers to the format for which information is stored, prior to presentation. The difference between Instagram and Twitter is thus the visual and textual mode, respectively. As such, Instagram is poised to advance research on how visuals aid social movements when held in comparison with the textual mode. We are particularly interested in two dimensions: framing and opinion leadership. First, the visual modality of engagement:through photos and video: demands a different form of participation than through text. Per the adage "a picture contains a thousand words," visuals are dense and powerful message holders. Studies have shown visual messages are more temporally efficient and amplify the affective reaction and reinforce textual content. Beyond photos, injustice symbols can serve as a force that extends political efforts beyond local, even national, accounts.
More critically, this form of engagement bears many similarities to the traditional relationship between press and protest. Long-standing work has shown institutional news media portrayals delegitimize collective action. As conceptualized, images of protest are distributed by legacy media sources. Protestors in turn sought their attention through disruptive or combative tactics, which led to condemnation, and thus delegitimization. The participation of citizen journalists, theoretically, would remove this necessity, and recent scholarship explores this possibility. A recent experimental study finds visual frames important for increasing support and identification toward protesters.
Apart from the modality itself, the legacy media has traditionally occupied opinion leadership, and this flow of information through elites has garnered significant attention since the proposition of two-step flow from more than half a century ago. While research that intersect visuals in social movements on digital media remains sparse compared to textual analysis, there are still numerous studies. For instance, Neumayer and Rossi (2018) analyze images recently studied photos and videos of Twitter during the Blockupy protests in Frankfurt. However, they found reinforcement in regard to the politics of visibility, where institutional and official accounts still garnered the most attention (retweets). Instagram, with its less-public interface and limited hyper-linkage could poise as a counterpoint to Twitter's organizational dynamics.
While offline events-such as widespread protests-certainly also occurred, contemporary social movements are coordinated significantly through online spaces, such as social media. Various reports have highlighted the importance of Instagram. If our assumption that a) online social platforms are important to protest organizing and b) Instagram has taken an outsized role compared to prior movements and Twitter, then answers to these questions can address how solidarity can be in part attributed to shifts in framing and shift in opinion leaders found on Instagram.
As such, our investigation into Instagram offers rich comparative insight toward a) how the modality of engagement shifts frames of legitimacy, and b) the nature and role of emergent opinion leaders. We offer the following research questions and hypotheses:
Characterization: What were the temporal trends of June 2020's second wave of the Black Lives Matter movement, in terms of frequency, geography, and textual content?
Injustice Symbols and Legitimization: What were the top shared images on Instagram, and how do they semantically, affectively, and symbolically function?
Network and Opinion Leaders: Do the central actors and communities that emerged on Instagram reinforce the institutional media, or are they conducive to grassroots connective action? | null | Not supported with pagination yet | null |
PMC7201439_01 | Male | 4 | A 4-year-old boy presented with an intermittent fever for two weeks, cold symptoms, and loss of appetite. On physical examination, the child was found moderately dehydrated, tachycardic, and tachypneic and demonstrated a hepatosplenomegaly. His peripheral blood tests showed 10% myeloid blasts with WBC (16.2 x 109/L), severe anemia (Hb 2.8 g/dL), and thrombocytopenia (platelets 6 x 109/L), as well as a monocyte count of 4.61 x 109/l. A bone marrow aspiration was performed and revealed 10% of myeloid blast cells. The patient was started on an induction regimen with cytarabine and rasburicase. Less than 24 hours after initiation of therapy, he developed fever and progressive respiratory failure. A chest X-ray showed a bilateral pulmonary infiltration. In the context of suspected sepsis, the broad-spectrum antibiotic treatment was started. Echocardiography showed signs of severe pulmonary hypertension (PHT). Despite inotropic support, mechanical ventilation, and early antibiotics, the patient deteriorated and was referred to our tertiary pediatric intensive care unit.
At arrival, his general condition was critical with signs of multiorgan failure and severe lactic acidosis. The neurological status appeared to be normal. His acute respiratory distress syndrome necessitated high-pressure ventilation (Figure 1(a)). Despite optimized conventional therapy, he rapidly developed cardiogenic shock. A veno-arterial (VA) ECMO was initiated via neck vessel cannulation, and immediate improvement in oxygenation was achieved (Figure 1(b)). ECMO flow was stable between 1.2 - 1.7 L/min during the whole support period. ACT was kept at 180 seconds. No major bleeding complications occurred. Mechanical ventilation was continued, and PHT therapy was initiated with sodium nitroprusside and nitrous oxide. First improvements in ventilation were observed on day 12 on ECMO support. On day 18, VA ECMO could be converted to a veno-venous (VV) ECMO by using a dual lumen Avalon ELITE cannula (MAQUET Cardiopulmonary AG, Germany) (Figure 1(c)). After seven days of VV ECMO support, the patient was successfully weaned on day 25 (Figure 1(d)). The neurologic assessment of the patient was limited during ECMO support due to pharmacological sedation. Following further hemodynamic improvement and weaning of sedation, the patient demonstrated a quadriplegia, global aphasia, and conjugate eye deviation. Subsequent MRI diagnostic revealed a central pontine myelinolysis. Certainly, it was caused by dyselectrolaemia with severe hypernatremia. Despite severe neurological impairment, his respiratory condition improved further, and 2 days later (day 30), he was extubated (Figure 2). Following extubation, a bilateral vocal cord paralysis of probably central origin was noticed.
Given the fast improvement of the patient, the initially paused chemotherapy with cytarabine was restarted early, thus allowing a cytogenetic remission on day 21 of ECMO support.
On day 34 of hospitalization, the patient was referred for evaluation for allogeneic hematopoietic stem cell transplantation to the transplantation center. A genetic testing revealed a KRAS and SETBP1 mutation. Given the severe neurological impairment, the decision was taken to proceed with bridging chemotherapy until improvement of the neurological status could be observed.
Five weeks later, the patient's clinical condition was normal with a remarkably improved neurological status and normalized vocal chord function. The patient could walk, even drive a 3-wheel bike, while his cognitive function was still impaired with a stammer requiring further speech and language therapy.
Six months later, he underwent allogeneic hematopoietic stem cell transplantation with a 10/10 HLA-matched unrelated donor donating bone marrow. The conditioning comprised fludarabine, targeted busulfan, and melphalan including rabbit ATG. He engrafted at day +26 and +23 with neutrophils and platelets of donor origin. The child was discharged home with minor cognitive impairment. | null | Not supported with pagination yet | null |
PMC9348830_01 | Male | 66 | A 66-year-old man presented and was admitted to the hospital with fever for 3 days and dyspnea for 1 day. The patient had been diagnosed with malignant pemphigus vulgaris and put on prednisolone, methylprednisolone and other immunosuppressive drugs for the past 9 years. On admission, he experienced respiratory failure. The absolute count of regulatory T lymphocytes was 156.67 cells/uL. The lactate dehydrogenase increased to 457 IU/L. The chest computed tomography (CT) scan revealed multiple flaky, fuzzy, and high-density opacities in both lungs (Figure 1A). The patient had been on steroids for a long time. He had obvious hypoxia and had never had any other respiratory symptoms or other infections. On respiratory examination, the patient did not have many rales in the lungs. The lung CT scan showed interstitial changes. PCP, usually treated with sulfonamides, was considered a possible etiology because of his immunocompromised state. Caspofungin is a good treatment of choice for Aspergillus infection, which could not be excluded. For other atypical pathogens, moxifloxacin should be added. Thus he was given trimethoprim-sulfamethoxazole (3 tablets, twice daily for 3 weeks), caspofungin (loading dose, 70mg infused over 1 hour; maintenance dose, 50 mg daily infused over 1 hour for 2 weeks), steroids, and moxifloxacin. Consequently, his respiratory function improved, and his body temperature dropped. PCP was confirmed 4 days later when cysts were found in the sputum (Supplementary Material).
Curiously, the follow-up chest CT suggested an enlarged consolidation in the left upper lobe (Figure 1A-C). The patient was admitted to the hospital again due to cough, expectoration, and intermittent fever. Bronchoscopy was performed for 3 times. Smears and culture of the bronchial brush, tracheal secretions, and bronchoalveolar lavage fluid (BALF) were performed. Lung tissue was taken for culture and pathological examination, and tuberculosis was diagnosed (Figure 2). Antituberculosis treatment (isoniazid 300 mg once daily, rifapentine 600 mg twice weekly, ethambutol 750 mg once daily, and pyrazinamide 500 mg thrice daily) was given for 2 months and later ethambutol and pyrazinamide were excluded. Antituberculosis treatment lasted for 1 year. Simultaneously, traditional Chinese medicine techniques were used to manage the patient's psychological well-being and improve mental state. After 3 years of follow-up, the clinical outcome was good (Figure 3). The patient's complete course is shown in Figure 4. | hiv-seronegative, immunosuppressed, mycobacterium tuberculosis, pcp, pneumocystis jirovecii, corticosteroids | Not supported with pagination yet | null |
PMC10408311_01 | Male | 4 | A 4-year-old boy presented with non-tender mass near the right lower molars. This had been incidentally noted by the kid's parents. The patient had no complaint of ache to the area. He was subsequently admitted to our hospital for further check. Laboratory blood examination of serum calcium, phosphorus, and parathormone levels was normal.
On physical examination, a smooth and non-tender mass was palpated in the right lower molars. The oral cavity checkup was not exceptional.
Computed tomography of the maxillofacial region showed a well-defined 25 mm x 16 mm osteolytic lesion affecting the right mandible; erosion of the cortex was present (Figure 1). The lesion was delicately excised using a piezosurgery and sent for frozen section that showed spindle cell, chronic inflammation, and macrophages. The gross specimen of the lesion showed whorled bundles of spindle-shaped fibroblasts (Figure 2), as well as foam cells, mingled with scant multinucleated giant cells, without any bone formation in the lesion. The lesion showed strongly and diffusely positive staining for SATB2 (Special AT-rich sequence binding protein 2) (Figure 3), focal positive for SMA (Anti-smooth muscle antibody), KP1 (Anti-CD 68 antibody), and ER (Estrogen Receptor) (Figure 4), and negative for H3.3G34W by immunohistochemistry. With respect to USP6 (Ubiquitin-specific proteases) break-apart fluorescence in situ hybridization, the result was negative (Figure 5). Whole exon sequencing for this lesion tested KRAS codon 12, codon 13, codon 61, and codon 146, which were all negative. The postoperative course was uneventful, the patient was followed for 15 months and continues to be asymptomatic. | non-ossifying fibroma, giant cell lesions, mandible | Not supported with pagination yet | null |
PMC3425151_01 | Female | 35 | A 35 year old woman, Hosp. No. - A 8332 came to our OPD on 05.08.05 with chief complaints of growth in the vulva - 2 month, bleeding from the growth - 1 month, irregular period - 6 month. She is P4+1. She chews betel nut and leaf. There is no family history of malignancy. No history of HTN, DM, TB. Tubectomy was done a year ago.
On examination, GC is fair, pallor +, no palpable LN. P/A- no organomegaly, others NAD, groin- NAD. Local examination of the vulva revealed a 8 x 5 cms pedunculated growth [Figure 1] having its thick pedicle of about 1.5 cm thickness hanging from the clitoreal region slightly on the left side [Figure 2]. P/S/V- cervix- NAD, vaginal walls- NAD, Uterus - N/S, mobile, adnexae free. P/R- NAD.
On investigation, Hb %- 9.8 gm%, ABO gr.- O +ve, RBS- 101 mg/dl, RFT-WNL, LFT-WNL. PAP Smear-NAD, CXR-NAD, USG (W/A) - NAD. Pre operative biopsy revealed Aggressive Angiomyxoma.
She was planned for wide excision of the vulval growth; accordingly it was done under SA on 18.08.05 [Figure 3]. On sectioning, the mass had focally infiltrating margins and a rubbery glistening grey/white surface [Figure 4]. Post operative period was uneventful, and patient was discharged on 27.08.05.
Post op HPE revealed Aggressive Angiomyxoma [Figures 5 and 6], cut margins negative. IHC studies revealed Desmin and Vimentin +ve, ER, PR +ve and S100 protein negative. She is under regular follow-up, and doing well for the last 5 years. | aggressive angiomyxoma, female perineum, myxoid neoplasm, myxoma | Not supported with pagination yet | null |
PMC9280111_01 | Female | 11 | A 11-year-old female child, who was apparently well 6 days before admission, complained of nonproductive cough, chest pain for 3 days, followed by diffuse abdominal pain, nonbilious vomiting, and shortness of breath for 3-day duration.
Further history revealed no personal or family history of recent infection. The child denied any history of fever, rash, blisters, jaundice, and trauma. She had dysuria, but there was no history of oliguria or hematuria. There was no neurological history contributing to the diagnosis. She had neither weight loss, nor history of contact with tuberculosis. She was previously fit and well and vaccinated as per the national immunization schedule. At the time of presentation, she was hemodynamically unstable with tachycardia (heart rate: 147/min), hypotension (blood pressure: 82/54 mmHg), and had moderate respiratory distress (respiratory rate: 32/min). She was resuscitated as per the standard Pediatric Advanced Life Support guidelines. An emergency bedside two-dimensional (2D)-echocardiogram [Figure 1a and b] demonstrated a massive pericardial effusion with cardiac tamponade along with bilateral pleural effusions. Her chest X-ray demonstrated cardiomegaly and bilateral pleural effusions [Figure 2a].
She underwent an emergency pericardiocentesis under ketamine sedation, and we drained around 600 ml of hemorrhagic fluid. She also had bilateral chest drains placed for bilateral pleural effusions [Figure 2b]. The right-sided chest drain had drained around 1000 ml hemorrhagic fluid, whereas the left-sided chest drain had 900 ml drain in 24 h. A 2D echocardiogram postpericardiocentesis demonstrated the resolution of pericardial and pleural effusions and moderately impaired left ventricular function with an ejection fraction (EF) of 35% raising the possibility of myopericarditis.
The child was managed with supportive care including oxygen therapy, intravenous (IV) antibiotics, oral Ibuprofen, and IV Milrinone. She was commenced on steroids (IV methylprednisolone 30 mg/kg for 3 days followed by tapering dose of oral prednisolone for 2 weeks) in view of suspected underlying myopericarditis. She showed clinical improvement after 48 h of admission, and both the pleural and pericardial drains were removed after 5 days of intensive care unit admission. Her chest X-ray at discharge demonstrated normal cardiac contours and the resolution of bilateral pleural effusions [Figure 2c]. Workup of pleural fluid showed a packed cell volume of 20%, neutrophils of 85% with no evidence of malignant cells, and investigations for tuberculosis or any other bacterial etiology were negative. Investigations including computerized tomography chest and collagen profile were negative for malignancy and connective tissue disorders, respectively. However, the workup for possible viral etiology revealed a positive IgM titer for Coxsackie B viral infection. She made a full recovery and was discharged after 9 days of hospitalization. At discharge, her left ventricle showed normal function with an EF of 65%, and she had no recurrence of pericardial effusion during follow-up. | coxsackie b virus, hemorrhagic pericardial effusion, hemorrhagic pleural effusion, pericardiocentesis | Not supported with pagination yet | null |
PMC5843970_01 | Female | 65 | A 65-year-old previously fit and well lady presented after collapsing with a sudden onset severe headache and nausea. She then developed a dense left hemiparesis and her GCS dropped to 6 (E1 V1 M4). Upon arrival at the emergency department she was intubated, and a computed tomography of the brain (CTB) revealed a 17 mm deep right acute-on-chronic SDH with 12 mm of midline shift. She had no history of trauma, did not take any regular medication (nor any sporadic doses of aspirin or nonsteroidal anti-inflammatory drugs in the months prior to admission), consume excess alcohol, or have any other cause for coagulopathy. She underwent a CT angiogram which did not show a vascular abnormality.
The patient underwent emergency craniotomy and evacuation of the SDH. Excellent hemostasis was noted during this operation. Postoperatively, she was admitted to neurointensive care where she remained intubated. After 4 hours, her GCS motor score dropped from 5 to 1 with equal and reactive pupils so she underwent a further CT scan. This showed a reduction in the size of subdural clot with persisting midline shift which was managed conservatively; 6 hours later her GCS had normalized and she was subsequently successfully extubated. However, in the next 2 to 3 hours, she became very drowsy and was no longer moving her left arm. A further CTB showed re-hemorrhage and she was taken back to theatre for repeat craniotomy and evacuation of SDH. This time her scalp was found to be "very oozy" with both subdural and extradural hematoma. Intraoperatively, a thromboelastogram (TEG) was performed, and the results (R 2.8, angle 34.4, and MA 19.6) were consistent with impaired platelet functioning. Based on these results, she was given 1 g tranexamic acid, 1 pool of platelets, and 2 units of cryoprecipitate. Her INR, APTR, and fibrinogen remained in the normal range throughout admission.
Eight hours post procedure she was awake and obeying commands. Postoperatively, she received a further 1 g tranexamic acid, 2 units of FFP, and 10 mg vitamin K to cover subgaleal and subdural drain removals.
Further questioning into her history revealed that she had been taking Red Clover for the preceding 8-10 years (364 mg per day of standardized Red Clover extract containing 40 mg isoflavones, as recommended by the manufacturer) as an over-the-counter herbal preparation for relief of menopausal symptoms. Red Clover was deemed to be a causative factor in her SDH and subsequent re-bleeding based on the abnormal TEG report showing impaired platelet functioning and absence of trauma or any other cause for coagulopathy.
A final CTB showed an improved SDH and reduced mass effect. Clinically, she was GCS 15 with a very mild right hemiparesis. She was transferred to her local neurosurgical unit for rehabilitation on postoperative day 3. At 6-month follow-up, she had returned to baseline functioning with no residual neurology. | herbal supplements, red clover, subdural hematoma | Not supported with pagination yet | null |
PMC3590287_02 | Male | 9 | The patient was a 9-year-old male referred at the age of six for orthopedic assessment because of right leg pain and gait disturbance. He was the eldest child of non-consanguineous parents. Maternal family history was unremarkable except for a second degree uncle with pes cavus. The parents were both healthy and the father reported that he was macrosomic at birth. Pregnancy was full term and delivery was uneventful. The patient's birth weight was 4270 g (>90th percentile), length 53 cm (>90th percentile). The patient's medical history revealed a mild delay in motor milestones, he started walking at 18 months. Speech acquisition was also delayed. At the age of 8 years, neurological examination confirmed motor awkwardness, psychomotor delay, and gait disorder (walking on toes and pes cavus). Cerebro-spinal MRI revealed swelling at D8-L1, hyperintense on T2-weighted images with irregular enhancement, ascribable to expansive intramedullary lesion without CNS dissemination. Bilateral dysgenesis of the lateral semicircular canal of the inner ear was also associated. Abnormalities of gyration and abnormal foliation of some sectors of both cerebellar hemispheres were also observed. The patient underwent to biopsy. Histological examination identified a pilocytic astrocytoma (WHO grade I). At physical examination, mild facial dysmorphisms was observed. The child presented low and flat forehead, very arched eyebrows, small nose with large filter, small mouth, downslanting and curled ears, short neck. Additionally, the child had small hands with flared distal phalanges, "drumstick-like"), and cafe-au-lait spots on the back. Auxological examination revealed mild obesity. The parents gave written informed consent for genetic studies.
Array-CGH analysis, performed on the proband, revealed the presence of two deletions of chromosome 22 and a duplication of chromosome 15. The extent of the deletion at 22q11.21 band (large deletion) was 1.417 Mb (chr22:18,894,835-20,311,763) (Fig. 4, A-B) while the second deletion at band 22q11.23 (short deletion) (chr22:25,664,618-25,911,651) spanned 247 Kb (Fig. 4, C). Furthermore, the patient carried a duplication spanning 484.3 Kb (chr15:100,051,182-100,996,155) at q26.3 band of a chromosome 15 (Fig. 4, D). The short deletion of chromosome 22 and the duplication of chromosome 15 were inherited from his apparently normal father, while his phenotypically normal sister inherited only the short deletion. His mother had no abnormalities (Fig. 3, B). The large deletion of chromosome 22 was described as proximally nested 1.5 Mb deletion harboring TBX1 and is present in 7-14% of DiGeorge/Velo-cardio-facial patients (DGS/VCFS). FISH experiments confirmed the presence of the short deletion on the metaphases of the father and showed that the large and short deletions were both on the same chromosome 22 on the metaphases of the son (Fig. 2, C). | null | Not supported with pagination yet | null |
PMC6800913_01 | Female | 50 | In July 2006, a 50-year-old female presented to the dermatology clinic with a complaint of painful skin changes in her bilateral lower extremities. She was employed as a dog groomer, and her past medical history was significant for type 2 diabetes mellitus with insulin requirements, gastritis, and severe valvular heart disease affecting the tricuspid, mitral, and aortic valves with surgical repair of the aortic valve. On physical examination, the patient was found to have linear, hyperpigmented macules on the bilateral lower legs with foci of scarring and ulceration. Two biopsies of the proximal and distal left lower leg suggested livedoid vasculopathy pending clinical correlation. She was additionally found to have an elevated antithrombin 3 activity of 124 (reference range 70.0-120.0), which is strongly suggestive of an underlying prothrombotic component to her condition. At this time, therapeutic options for livedoid vasculopathy were considered and offered to the patient.
The patient's preferences and past medical history presented several obstacles to treatment of her LV. She repeatedly refused anticoagulant therapy because her husband had previously had issues with the diet restrictions and INR monitoring mandated by the use of warfarin. Antiplatelet agents were avoided due to her history of severe gastritis that was onset prior to our management of her LV. Intravenous immunoglobulin was considered; however, the patient could not afford the co-pay and her cardiologist recommended against IVIG due to the risk of these hyperosmolar preparations causing fluid overload in this patient with severe valvular heart disease. The patient was eventually started on an acceptable treatment regimen consisting of oral dapsone 100 mg once daily and prednisone 10 mg once daily, with the addition of doxepin or tramadol for intermittent pain control. With these medications, she achieved intermittent remission of her LV for several years.
In 2015, she began having intermittent, painful flares of her LV which were managed by increasing her dapsone to 150 mg once daily and increasing her prednisone to 20 mg. She sometimes required burst doses of 60 mg once daily. Attempts to wean her prednisone back down to 10 mg were rarely successful, and this dosing became an ongoing concern in 2018 when she began having severe hyperglycemic episodes which resulted in a brief hospitalization. Her insulin delivery was also switched to a pump system.
In the fall of 2018, she presented to dermatology during an acute LV pain flare and was coincidentally found to have an erythematous papule at the right dorsal forearm, which she attributed to a possible insect bite or a scratch from her dog. Three weeks later, she reported worsening of this right forearm lesion as well as new onset of two painful, ulcerative lesions on her right thigh and right forearm. On examination, the right dorsal forearm was now found to have two firm, tender erythematous papulonodules, one with central ulceration (Figure 1). The right lateral thigh was found to have a single purpuric patch with central ulceration and necrosis (Figure 2). The bilateral forearms additionally had a few scattered linear superficial abrasions consistent with animal scratches. No other new lesions were found on examination. She perceived these lesions as dissimilar from her typical LV, but at this time mycophenolate mofetil 500 mg twice daily was added for her ongoing LV flares. A 4 mm punch biopsy of a right dorsal forearm lesion revealed suppurative, granulomatous inflammation in the deep reticular dermis with demonstration of acid-fast organisms on AFB stain (Figure 3).
The patient was subsequently evaluated for systemic mycobacterial disease. A chest X-ray showed no evidence of pulmonary mycobacterial disease. Laboratory testing was negative for HIV, AFB blood culture, and Tb QuantiFERON. Her CRP was elevated at 13.3, and the CBC revealed a mild leukocytosis of 13.00 with mild neutrophilia of 79.4%, elevated absolute neutrophils (10.3), and absolute immature granulocytes (0.4).
The state Department of Health identified rapidly growing Mycobacterium abscessus/chelonae on her right forearm tissue culture. Susceptibility testing and treatment guidance was subsequently provided by a national center specializing in mycobacterial consultation. Susceptibility results revealed resistance to cefoxitin, doxycycline, sulfamethoxazole-trimethoprim, and amoxicillin-clavulanic acid and sensitivity to clarithromycin, azithromycin, linezolid, imipenem, and amikacin. There was also intermediate sensitivity to ciprofloxacin and moxifloxacin.
The physicians of the Dermatology and Infectious Disease departments then coordinated management of her simultaneously flaring livedoid vasculopathy and disseminated cutaneous mycobacterial infection. The patient had a PICC line placed for her antimycobacterial regimen which consisted of oral azithromycin 250 mg once daily, intravenous imipenem 500 mg every 12 hours, and finally intravenous amikacin 12-15 mg/kg on Mondays, Wednesdays, and Fridays. Additional treatment with fluoroquinolones was not considered because she had a previously documented allergic reaction of a blistering skin eruption. This regimen was initially to be continued for a total of 8-12 weeks depending on her response, followed by oral azithromycin monotherapy for at least an additional 6 months. Lifelong suppression with oral monotherapy is also being considered pending her clinical response and tolerance to azithromycin.
At 10 weeks of treatment her physical exam revealed improvement of the ulcers of the right forearm, right thigh, and left foot. However, she also developed three new left knee ulcers consistent in appearance with her other mycobacterial lesions. For this reason, her current regimen is to be continued until her follow-up appointment for PICC line removal at 17 weeks of treatment. At that time, she will transition to daily maintenance therapy of oral azithromycin if she demonstrates adequate improvement in the existing lesions.
Meanwhile, the treatment of her livedoid vasculopathy was optimized by the dermatology team. Her prednisone was tapered down to 10 mg once daily, and both the dapsone and mycophenolate mofetil were discontinued. She has since been started on warfarin 1 mg once daily. This low dose was chosen due to reports of supratherapeutic INR from interaction of warfarin and azithromycin. Her LV was found to be stable at her follow-up appointment 9 weeks after initiating and continuing the same dose of warfarin. | null | Not supported with pagination yet | null |
PMC7513585_01 | Male | 41 | Research ethics approval was obtained from the Research Ethics Boards of Simon Fraser University, Fraser Health Authority, and the National Research Council of Canada. As co-investigators and co-authors, Captain (retired) Trevor Greene (TG) and his wife Debbie Greene (DG) continue as collaborators in a combined PT/functional neuroimaging longitudinal case study that began in 2010. D'Arcy et al. provided a detailed case description. In summary, TG was 41-years-old, right-handed, and university-educated when he was on tour in Afghanistan in 2006. He was leading a meeting with Shinkay village elders on March 4, 2006, to help provide access to basic needs. He and the other soldiers had removed their helmets and laid down their weapons as a gesture of respect. At this point, a 16-year-old male pulled out an axe and swung it with two hands from above his head into the top of TG's skull. The act signaled a Taliban attack.
The axe blow resulted in a severe open TBI with a deep penetrating injury along the midsagittal plane, extending from the frontal to the parietal lobes, with greater right frontal and left parietal damage relative to the sagittal suture. Primary motor, premotor, primary somatosensory, and superior parietal areas were affected. The depth of the injury extended from the cortex inferiorly to the lateral ventricle, affected anterior cingulate gyri, corpus callosum (body and genu), and surrounding white matter tissue.
Immediately afterward, TG underwent emergency care and was medevacked to Kandahar Airfield, where he was transferred to Germany for neurosurgical treatment and induced into a medical coma at the US Army Landstuhl Regional Medical Centre. TG was subsequently transported back to Vancouver General Hospital (Vancouver, BC, Canada), with an initial prognosis of permanent vegetative state. During acute care, he emerged from the coma and gradually recovered full consciousness over the following 18-months. He was then admitted to the Halvar Jonson Centre for Brain Injury Centre (Alberta, Canada) for a 14-month intensive rehabilitation program.
Since then, TG has managed severe physical movement disabilities through intensive daily rehabilitation. He has returned to his career as a journalist/writer after retiring from active duty, is a published author of several non-fiction books, speaker (including TEDx), and holds honorary doctorates along with other notable distinctions. He has actively engaged in cognitive training while managing on-going post-traumatic stress disorder (PTSD). For TG, on-going PTSD challenges include night terrors and being unable to sit with his back to a door. For a full description of TG and DG's rehabilitation journal, see their book entitled: "March Forth: An Inspiring True Story of a Canadian Soldier's Journey of Love, Hope, and Survival" (Greene and Greene,). TG's rehabilitation objectives continue to focus on recovering walking abilities along with all other related impacts. As a former elite rower, he applies intensive daily training together with mental imagery to continue to push the limits of his recovery.
The objective of the longitudinal case study has been to monitor TG's recovery using multimodal functional neuroimaging (i.e., fMRI, MEG, and EEG). In the first phase (D'Arcy et al.,), fMRI was used four times a year over 3 years (12 times total) to monitor motor activation recovery. In parallel to clinical measures of movement recovery, a significant 5-fold increase in lower limb motor activation was observed. Of note, TG also showed significantly higher mental imagery activity (imagined rowing) relative to a matched control in the same active regions. The findings demonstrated neuroplasticity-related recovery well beyond conventional limits of 6-months to 1 year (6+ years at the time of the study).
The second phase incorporated assistive device technologies following an extended plateau in rehabilitation progress (D'Arcy et al.,). Following various assistive device trial evaluations, the study phase began in 2018 with the specific goal of investigating whether non-invasive neuromodulation, when paired with continuing physical rehabilitation, could help overcome the plateau of the recovery beyond 12-years post-injury. TLNS through the PoNS was highlighted in the book The Brain That Changes Itself (Doidge,). To evaluate whether PT + TLNS could facilitate further recovery, we collected clinical, EEG, and MEG data during a 1-year PT only plateau period (i.e., baseline) and a 14-week PT + TLNS period (i.e., treatment). In a parallel study of motor function, PT + TLNS treatment led to clinically significant motor ability improvements with corresponding significant changes in both EEG and MEG activation suggesting network-level neuroplasticity effects in motor control function (D'Arcy et al.,). The present study focuses on the associated cognitive changes. | brain vital signs, case report, electroencephalography (eeg), neuroplasticity, post-traumatic stress disorder (ptsd), translingual neurostimulation (tlns), traumatic brain injury (tbi) | Not supported with pagination yet | null |
PMC10041614_01 | Male | 51 | A 51-year-old male presented to the emergency department with pain and facial swelling in the right cheek, which had been ongoing for 2 to 3 months. In the emergency department, a computerized tomography (CT) scan showed inflammation in the right maxillary sinus with mucosal thickening. A complete blood count (CBC) with differential and comprehensive metabolic panel (CMP) were both within normal limits. He was started on a 10 days course of oral antibiotics and fluticasone nasal spray.
Nine days later the patient presented to his primary care physician with concern for worsening right facial swelling, despite being on an antibiotic, and represented to the emergency department. The patient was started on vancomycin, piperacillin/tazobactam, and dexamethasone. A magnetic resonance image (MRI) of the brain was obtained and revealed a mass within the right infratemporal and pterygopalatine fossae extending into the inferior right orbital fissure along V2 and the vidian nerve, causing concern for potential malignancy (Figures 1 and 2).
He was transferred to a tertiary care center for evaluation and management. On physical examination showed diminished sensation at the right maxillary division of the trigeminal nerve (V2) but no double vision. His right face was swollen but not fluctant and he had trismus. The patient was taken to the operating room for biopsy. The sinonasal mucosal surface all appeared normal. The posterior wall of the maxillary sinus was removed to allow access to the mass, the internal maxillary artery appeared necrotic. Histological assessment demonstrated multiple arteries with partial to complete luminal obliteration associated with loosely formed non-necrotizing granulomas, consistent with granulomatous vasculitis (Figure 3). Serum studies were positive for antinuclear antibodies (ANAs) but were negative for anti-neutrophil cytoplasmic antibodies (ANCA).
The patient was evaluated by an infectious disease specialist and was negative for human immunodeficiency virus (HIV), hepatitis B and C, Mycobacterium tuberculosis, and other diverse opportunistic organisms. The patient was referred to a rheumatologist and nephrologist. Urine sediment showed 1 to 2 dysmorphic red blood cells (RBCs) per high-power field and 3 to 5 monomorphic RBCs per high-power field, concerning for renal involving vasculitis. The patient was diagnosed with ANCA-negative granulomatosis polyangiitis and was started on immunosuppressive treatment with prednisone and mycophenolate. At his 1-month follow-up with a nephrologist and otolaryngologist, the patient reported improved symptoms with decreased facial swelling, the ability to open his mouth, and improved sensation on the right side of his face. An informed written consent was obtained from the patient for publication of this case report. | gpa, granulomatosis with polyangiitis, wegner, granuloma, skull base mass, vasculitis | Not supported with pagination yet | null |
PMC3763677_01 | Male | 30 | A 30-year-old male was involved in an automobile accident. He was at the driver's seat and did not wear a seatbelt. During admission, his primary complain was back pain.
Upon admission, palpation revealed tenderness at the thoracolumbar region. His neurologic examination was normal with no motor or sensory deficit of the lower and upper limbs. Ultrasonography was performed to exclude blunt abdominal concomitant injuries. Simple radiographic evaluation of the cervical spine, thorax and pelvis did not demonstrate associated injuries. Radiographs (Figs. 1, 2) and CT scans (Fig. 3) of the thoracolumbar spine revealed a transverse fracture at the level of T12. The CT scan further demonstrated a split of the posterior elements, progressing anteriorly into the vertebral body. Conservative treatment was decided and the patient, following an initial period of bed rest, was mobilized wearing a thoracolumbar orthosis. Appropriate antithrombotic prophylaxis with low molecular weight heparin was administered, until full ambulation was achieved. The patient was followed up with serial thoracolumbar radiographs for the first 6 weeks of ambulation and later on, in one month intervals. The thoracolumbar orthosis was applied for three months. At the 5th month follow-up, he was pain free with radiographic signs of mild vertebral kyphosis. Upon completion of treatment, he returned successfully to pre-injury levels of daily activity, without any impairment. | chance fracture, seatbelt injury, thoracic spine | Not supported with pagination yet | null |
PMC8476786_01 | Male | 50 | A 50-year-old male with end-stage renal disease received a left kidney transplant from a deceased male donor who died in a motor vehicle accident in March 2013. After renal transplantation, the patient received a maintenance immunosuppressive regimen consisted of tacrolimus (3.5 mg, bid), mycophenolate mofetil (360 mg, bid), and prednisone (4 mg, qd). At 11 months postoperatively, 1+ to 2+ proteinuria was found on a routine urine examination. The proteinuria was relieved after treatment with Tripterygium glycosides tablets (10 mg, bid). At 15 months postoperatively, the patient developed BKV viruria with a urinary viral load of 1.25 x 107 copies/mL (normal range for reference, <5,000 copies/mL). The viral load was undetectable after the dosage of tacrolimus was reduced to 1.5 mg BID and treating with immunoglobulin (infusion). In April 2016, the patient had a chronic rejection reaction and the 24-h urinary protein quantity increased to 1.13 g/24 h. To maintain the allograft function and suppress proteinuria, the corresponding treatment regimen was methylprednisolone (40 mg) combined with cyclophosphamide (0.2 g) intravenous drip for 3 days/month. After three courses of treatment, the 24-h urinary protein quantity decreased to 0.56 g/24 h. The patient has had recurrent symptoms of UTI such as frequent and urgent urination without obvious inducement since June 2016. Regular outpatient review of urinary examination revealed leukocytes fluctuating from 1+ to 3+. E. coli was detected in the midstream urine culture and intravenous cefoperazone sodium sulbactam (1.5 g, 1/12 h) was given for 1 week. In September 2018, the patients came to the hospital because of cough for 1 day. mNGS of the alveolar lavage fluid indicated pneumosporidiosis and blood tested positive for herpes simplex virus. The pulmonary infection resolved after treated with compound sulfamethoxazole tablets (480 mg, bid). Besides, serum creatinine decreased from 305 mumol/L to 245 mumol/L. However, BKV viruria relapsed with the urinary viral load fluctuated from 2.84 x 105 copies/mL to 3.81 x 107 copies/mL. The immunosuppressive regimen was adjusted to tacrolimus (1.5 mg, bid), mycophenolate sodium enteric-coated tablets (180 mg, bid), and prednisone (4 mg, qd). In November 2018, the patient's serum creatinine was 197 mumol/L, and color Doppler examination of the transplanted kidney and renal vessels showed no significant abnormalities. Regular color Doppler ultrasound examinations of the allograft and transplanted kidney vessels were performed every 6 months after transplantation, all showing neither significant abnormalities nor transplanted kidney masses until the current admission. The patient had not undergone an allograft puncture biopsy within 3 years after renal transplantation. His postoperative serum creatinine level was 190 mumol/L. And the postoperative glomerular filtrate rate (GFR) was summarized in Figure 1. The patient was admitted to hospital in May 2019 because of frequent and urgent urination. The clinical course of the patient is summarized in three phases according to the disease progression.
In May 2019, the patient was admitted to hospital for recurrent frequent and urgent urination. On admission, his serum creatinine level was 229 mumol/L. Ultrasound and PET/CT showed solid space-occupying lesions in the upper pole of the transplanted kidney and bladder, prostate and seminal vesicles (Figures 2A1-H1). Biopsy of the graft kidney and bladder lesions revealed cryptococcal granulomas, with cystoscopy results provided in Supplementary Figure 1. Extended-spectrum beta-lactamase positive E. coli was cultured from both renal graft tissue and midstream urine. To rule out systemic cryptococcosis, a lumbar puncture was performed to collect CSF and CSF opening pressure measured. Cell counts and biochemical parameters in CSF, and CSF opening pressure were normal. Ink staining and Cryptococcus antigen detection of CSF were both negative. No significant abnormalities were observed in brain MRI and chest CT scans. Based on the imaging and pathological findings, the patient was diagnosed with cryptococcoma and malacoplakia of the genitourinary system including transplanted kidney, bladder, prostate and seminal vesicles (Figures 2A1-H1 and Figure 3), accompanied by an UTI of E. coli. The patient was treated with meropenem (0.5 g twice a day), and fluconazole (50 mg twice a day) combined with flucytosine (0.5 g twice a day). Meanwhile, immunosuppression was reduced by conversion from tacrolimus (1.5 mg twice a day) to cyclosporine (100 mg twice a day). Additionally, cyclosporine was later adjusted to 75 mg BID after use of the antifungal drug, fluconazole, which can affect the concentration of cyclosporine. The concentrations of immunosuppressants from renal transplantation to transplant nephrectomy were shown in Supplementary Figure 4. After 10 days' treatment, his renal function improved and the serum creatinine decreased from 229 to 185 mumol/L. In July 2019, his chest X-ray showed a soft tissue shadow in the left hilum, which was diagnosed as pulmonary tuberculosis by bronchoscopy; and the patient was thus treated with isoniazid (300 mg, once daily), ethambutol (750 mg, once daily) and levofloxacin (250 mg, once daily).
Follow-up ultrasound and FDG-PET/CT examination in August 2019 showed that although the area of bladder lesion was significantly reduced from 4.7 x 4.1 x 5.5 cm to 3.8 x 2.3 x 3.6 cm, the lesion in the transplanted kidney was enlarged from 4.3 x 4.9 x 4.1 cm to 6.0 x 4.0 x 4.7 cm, with the cryptococcoma and malacoplakia in the upper pole of the transplanted kidney protruding into the adjacent Gerota's fascia (Figures 2A2-H2). Fluconazole was given at 100 mg BID against cryptococcal infection while the anti-Escherichia coli as well as the anti-tuberculosis regimen was maintained, as described in Phase I. The net immunosuppression status of an individual is influenced by the immunosuppression regimen and individual susceptibility and can be assessed by immunosuppressive drug concentrations, peripheral blood leukocyte counts, lymphocyte counts, and viral infection conditions. The peripheral blood leukocyte and lymphocyte counts of this case were lower compared to the average level during uninfected period (Supplementary Figure 5). The presence of pneumosporidiosis, herpes simplex virusemia, and BKV uremia were all suggestive of a low net immunosuppressive status. To stop the disease progression, the patient received a transplant nephrectomy 1 week later.
After transplant nephrectomy, the patient's immunosuppressants were discontinued. He was on dialysis three times a week. The anti-tuberculosis treatment was changed to isoniazid (300 mg, once daily), ethambutol (870 mg, once every 2 days) and levofloxacin (250 mg, once every 2 days), while the anti-Cryptococcus treatment was changed to fluconazole (100 mg, twice a day) and flucytosine (0.5 g, three times a day). After 2 months, a follow-up FDG-PET/CT showed that the lesions in the urogenital system and lung were significantly reduced compared with the last examination (Figures 2A3-H3). Six months post-transplant nephrectomy, the lesions in the genitourinary system were eventually eliminated (Figures 2A4-H4).
Gross inspection of the transplanted kidney showed that the resected transplant volume was 11 x 9.0 x 5.0 cm with its capsule closely adherent to the surrounding fat. A solid yellow mass of 6.0 x 4.0 x 4.7 cm was found in the upper pole renal parenchyma without breaking through the renal capsule. Another 5.0 x 2.5 x 2.5 cm yellow mass was found in the hilar sinus fat (Figure 3F). The pathological changes of the transplanted kidney lesions were consistent with Cryptococcus infection (Figures 3A-E).
Metagenome sequencing revealed that only two and four sequences of C. albidus were respectively detected in the CSF and feces specimens in Phase I. Two sequences of C. albidus were detected in the CSF specimen in Phase II. After transplantation nephrectomy, withdrawal of immunosuppressants and anticryptococcal therapy for 2 months, one sequence of C. albidus was detected in the feces specimen but no sequence in CSF in Phase III. All sequences were typed as [Cryptococcus] albidus var. albidus strain NRRL Y-1402 (Table 1). The type of the E. coli detected in urine and granuloma of the allograft in Phase I and Phase II was E. coli 09-02E (Table 1).
In order to understand the interactions between E. coli and Cryptococcus, we cocultured the two microorganisms in vivo. After an 8-h co-culture of E. coli 09-02E filtrate and Cryptococcus neoformans JEC21 (ATCC@96910) in vitro, the Cryptococcus counts in the control group (without E. coli filtrate) and the experimental group (adding 80, 160, 320, 640, and 1,280 mul E. coli filtrate, respectively) were respectively 2.46 +- 0.52 x 105/mL, 2.30 +- 0.57 x 105/mL, 2.83 +- 0.72 x 105/mL, 3.13 +- 0.76 x 105/mL, 3.09 +- 0.61 x 105/mL, and 2.60 +- 0.63 x 105/mL. Comparison between the control group and the experimental Group III (320 mul E. coli filtrate) and Group IV (640 mul E. coli filtrate) showed a statistically significant difference (P < 0.05) (Supplementary Figure 2), indicating that E. coli at these concentrations may stimulate cryptococcal growth. | cryptococcus albidus, escherichia coli 09-02e, cryptococcoma, malacoplakia, metagenome sequencing, transplanted kidney | Not supported with pagination yet | null |
PMC8608600_01 | Male | 39 | A 39-year-old man diagnosed in childhood with cystic fibrosis (genotype 3849 + 10 kb leading to a C to-T change, homozygous) had been followed at a CF specialty clinic but had significant gaps between visits. The patient presented with a three-day history of dyspnea, productive cough, fevers, and hypoxia. Prior to hospitalization patient was on as needed oxygen at night, but had increased to 2.5 lpm at home over the past three days. Chest radiograph showed new bilateral perihilar infiltrates and laboratory evaluation showed leukocytosis (14 x 103 cells/muL). The patient had a history of colonization with methicillin-resistant Staphylococcus aureus and Pseudomonas aeruginosa, chronic sinusitis, obesity, and gastric reflux disease. Patient did not have history of pancreatic insufficiency prior to hospitalization. Three months prior to hospitalization, pulmonary function tests were FVC 3.60L/76% predicted, FEV1 2.18L/57% predicted, FEV1/FVC 60/75% predicted, FEV 25-75 30% predicted; oxygen saturation was 93% on room air. At that time patient was also diagnosed with thrush and tinea cruris. Prior to that visit there was a 3-year gap in care. At the time of hospitalization home medications included albuterol/ipratropium inhalation solution, azithromycin, docusate, dornase alfa, ergocalciferol, fluticasone/salmeterol inhalation powder, multivitamin, pantoprazole and tobramycin inhalation solution. The patient denied tobacco, alcohol, or recreational/injection drug use. He was in a monogamous relationship with a female partner. The patient denied history of having sexual contact with men. The patient also had no history of blood transfusions. Patient did report that a former female partner died of an unknown illness several years prior to his hospitalization.
The patient was admitted to the hospital with a presumed diagnosis of CF exacerbation. Initially the patient received 5 lpm of oxygen and oxygen saturation was 94%. Intravenous cefepime, tobramycin, vancomycin and standard CF therapies were initiated. Infectious Diseases and Pulmonary services were consulted. Evaluations for causes of acute hypoxic respiratory failure included bacterial, fungal, and mycobacterial sputum cultures, mycoplasma and coccidioides serologies, screening for connective tissue disease, urine legionella antigen, urine pneumococcal antigen, and respiratory viral panel, all of which were unrevealing. An echocardiogram showed normal left ventricular systolic function (LEVF) with pulmonary artery pressure of 37 mmHg. The patient's pulmonary condition worsened; on hospital day 4 he developed respiratory distress and sudden increase in oxygen requirement. A computed tomography (CT) scan of the chest with contrast showed extensive bilateral parenchymal ground glass opacities with peribronchial consolidation, read as more consistent with atypical pneumonia than with CF exacerbation, though pre-existing bronchiectasis made interpretation difficult. The patient was transferred to the intensive care unit (ICU) (see Fig. 1). On hospital day 5 a consultant suggested HIV testing, as well as a bronchoscopy, but the patient was initially unstable and the procedure was deferred. On hospital day 8, an HIV screen was positive and confirmed with Western blot; CD4+ cell count was 56 cells/muL and HIV viral load was 947,000 copies/mL. On hospital day 9, the patient underwent bronchoscopy; pneumocystis was identified by calcofluor stain from bronchoalveolar lavage. The patient was treated with sulfamethoxazole/trimethoprim 15mg/kg/day (based on trimethoprim component), prednisone taper, and antiretroviral therapy. The patient responded to treatment and did not require intubation; however, he developed a spontaneous pneumothorax, requiring multiple thoracostomy tubes (one with a Heimlich valve) and eventually was transferred to a specialty hospital for chemical pleurodesis. Screening tests for sexually transmitted diseases, tuberculosis, hepatitis, and toxoplasmosis were negative.
The patient is now followed by both the HIV and CF specialty clinics at our institution and he has remained stable with chronic oxygen requirement (2-3 LPM), improved CD4 cell count and an undetectable HIV viral load. No evidence of prior HIV screening was documented prior to hospitalization. His current female partner is not infected with HIV. | cystic fibrosis, hiv, pneumocystis pneumonia | Not supported with pagination yet | null |
PMC10082677_01 | Male | 5 | Youth patient A (YP-A) was a 17.5-year-old, Caucasian male with previous diagnosis of ASD from neuropsychological testing at age 8. At intake, he reported history of heavy binge use of alcohol (approximately 2 binge episodes per week, 6-8 standard units of alcohol per binge) and marijuana (5/7 days of cannabis use, unknown potency, but 1-2 joints per time). He had also been misusing cough medications daily (approximately 900 mg of Delsym; dextromethorphan per occasion) for the majority of days in the three months prior to the initial evaluation and reported stealing this product from a local pharmacy. YP-A and his parents also reported challenges in patient's emotional regulation typified by explosive outbursts with intermittent nonsuicidal self-injurious behavior (NSSIB) in the home. He was diagnosed with DSM-V autism level 1, alcohol use disorder - mild and cannabis use disorder - mild. He did not meet DSM-V criteria for other substance use despite evidencing problematic use patterns, a rule out of mood disorder was also given due to explosive behavior and intermittent NSSIB. Age of onset of first alcohol use was 16 years, cannabis 16 years, and dextromethorphan was 17 years. All DSM-V substance use diagnoses were given at the intake with assessed time period being the past 12 months. Family history was significant for alcohol problems on paternal side (although it was not known who specifically). Treatment goals focused on safety planning, communication skills, functional analysis of use, contingency management, and emotion regulation skill development.
YP-A was able to complete 9/12 youth modules (75% of modules implemented) over 11 sessions, over approximately a 5-month period; specifically, those focusing on psychoeducation about ASD and SUD, emotional identification and regulation skills training, sobriety sampling, and social and communication skills. The order of modules was altered at times to accommodate the patient's needs of the session, and modifications were made to abbreviate some modules or extend them over multiple sessions as the patient was only able to tolerate meetings that were less than 20 minutes in duration. The patient was able to track use of substances, set goals for reducing use of cannabis, avoid binge drinking, and set a 30-day sobriety sample from more risky cough syrup use. He also used emotion regulation skills and communication strategies to reduce anger outburst with his parents at home. The patient was also willing to engage with the psychiatrist for a trial of mood stabilizing medication by seeing the clinic psychiatrist.
Parent engagement was limited as his parents were well educated on ASD given the early diagnosis in childhood. Parents completed 5/7 modules (70% of modules implemented) with sessions focused on how to use contingency management and communication skills in order to enhance positive reward for improved or nonsubstance use behavior, reduced behavioral dysregulation (e.g., anger management-taking space, using communication skills more effectively, reaching out to therapist when in distress) as well as improving overall relationships within the home. A joint session via telehealth was unsuccessful due to YP-A feeling overwhelmed with the video-based format (implemented during the COVID-19 pandemic; thus, a combination of video visits were done) Despite limited parental engagement, this patient engaged consistently with therapy, started medication to target his mood, and was still smoking cannabis but fewer times per week (3-4 days per week). Importantly, he had not used cough medicine or engaged in binge alcohol use since starting treatment. Though not sober, engaging in less risky substance use was an important treatment outcome in this case. Regarding CGI outcomes incorporating both patient and parent therapist impressions, outcomes for substance use, YP-A initial CGI-S score was 5-6 (markedly/severely ill), and at the end 3 (mildly ill), with CGI-I indicating 2 (much improved). Regarding ASD symptoms, initial CGI-S score was 4 (moderately ill), and at the end 3 (mildly ill), with CGI-I indicating 2-3 (much improved/minimally improved). | null | Not supported with pagination yet | null |
PMC8266439_01 | Female | 18 | An 18-year-old female patient with no notable pathological history, consulted for chronic pain in the right iliac fossa evolving in a context of anorexia and weight loss of 10 kg in 4 months. On general examination, the patient was apyretic, conscious, and hemodynamically stable, and her conjunctiva was normally colored. The physical examination revealed a subumbilical mass mobile in both superficial and deep planes, undefined borders, and extending into the right iliac fossa; no hepatomegaly, splenomegaly, or lymphadenopathy was detected. Other systemic examinations were unremarkable. Her blood investigations were normal; her chest X-ray was also normal. The abdominal CT scan (Figure 1) showed two contiguous, well-limited, rounded formations with thick fluid content and thin, regular walls, they measured, respectively, 20 mm at the level of the right iliac fossa and 72 mm at the abdominopelvic level, under the umbilicus. The pelvic magnetic resonance imaging (Figure 2) showed a median intraperitoneal pelvic mass hyposignal on T1 and hypersignal on T2 and regular parietal enhancement after injection with mural nodules, it measured 77 x 81 x 80 mm; this mass was supravesical, coming into contact with the ileal intestines, the sigmoid, and the uterus behind it, keeping a separation line.
Laparotomy exploration showed a mesenteric mass close to the ileocaecal region, solid hyper vascularized with some ileocaecal lymph nodes (Figure 3); the appendix and the ovaries appeared normal; a monobloc resection of the mass with the satellite lymph nodes was performed without intestinal resection (Figure 4). The postoperative course was simple, and she was discharged home after 4 days in good general condition.
Microscopic pathologic examination was in favor of a necrotizing granulomatous lymphadenitis which was highly indicative of tuberculosis (Figures 5 and 6). Polymerase chain reaction (PCR) test of the specimen was positive for Mycobacterium tuberculosis.
Quadruple therapy for tuberculosis with isoniazid, rifampicin, pyrazinamide, and ethambutol was prescribed for a two-month initial phase of treatment followed by a four-month continuation phase of isoniazid and rifampicin. In patient's follow-up, general condition was good and no problem was reported. | null | Not supported with pagination yet | null |
PMC6886628_01 | Female | 55 | A 55-year-old Caucasian female with an active smoking history of 25 pack-years was referred to our pulmonology department due to complaints of chronic intermittent rhinitis and for vigilance of a 6 mm nodule on the chest CT scan in the right upper lobe. Imaging surveillance was maintained but 12 months after the initial chest CT scan, in addition to a stable 6 mm nodule on the right upper lobe, a heterogeneous and irregular 36 mm mass, with a central hypodense zone (RHS), was diagnosed on the apico-posterior region of the left upper lobe (Fig. 1)
The patient had no respiratory or constitutional symptoms and denied complaints suggestive of connective tissue disease. Her husband had been treated, 6 months earlier, for PT but her contact screening was negative. She had no prior relevant medical history and was being treated for her rhinitis with budesonide nasal spray 100 mcg and levocetirizine 5 mg.
The general examination was unremarkable, except for a pale nasal mucosa.
Laboratory analysis showed an increase in haemoglobin levels, 16.2 g/dl (normal 12-15 g/dl), probably due to the patient's smoking history, with no other relevant changes, particularly regarding inflammatory markers, which were normal. Due to her smoking history, a panel of tumour markers was requested (CEA, CA 19.9, CA 15.3, CA 125, CYFRA-21, NSE and SCC) which were also normal. Serologic tests for hepatitis B, C and HIV were negative and autoimmune antibodies (anti-CCP, ANAs and ANCAs) were within normal values. Pulmonary function tests revealed small airway obstruction with a normal diffusing capacity for carbon monoxide.
The case was discussed in a multidisciplinary team meeting. Given the lack of symptoms, the appearance of a relatively large mass over a short period of time (which made the diagnosis of cancer less likely) and the presence of the RHS on the chest CT scan, OP was considered the most likely diagnosis and initiation of corticotherapy was suggested.
Before starting corticotherapy, in order to rule out alternative causes and confirm the diagnosis, a bronchofibroscopy with bronchoalveolar lavage (BAL) and a percutaneous CT-guided transthoracic needle biopsy of the lung were performed. Pathological analysis of the lung biopsy revealed necrotic tissue. The BAL findings were nonspecific: a total of 63 ml (out of 150 ml) of opaque fluid was recovered with a total cell count of 494 cells/mcl with a macrophage predominance (94%). Microbiological and cytological analyses of the BAL fluid were negative for infectious or neoplastic disease. However, bronchial secretions collected during the bronchofibroscopy showed Mycobacterium tuberculosis (MT), susceptible to all first-line drugs (isoniazid, rifampin, pyrazinamide and ethambutol).
The patient had the final diagnosis of PT and not OP, as initially suspected.
She was referred to a specialised tuberculosis center and the recommended treatment for drug-susceptible PT was initiated. The patient completed a 2-month intensive phase with 4 drugs (isoniazid 300 mg/day, rifampin 600 mg/day, pyrazinamide 1,500 mg/day and ethambutol 1,200 mg/day) and is now completing the 4-month continuation phase with: isoniazid 300 mg/day and rifampin 600 mg/day. She remains clinically stable, asymptomatic and without adverse effects arising from the medication. A re-evaluation chest CT scan was performed and showed imagiological improvement, confirming the diagnosis of PT. | reversed halo sign, organising pneumonia, pulmonary tuberculosis | Not supported with pagination yet | null |
PMC9810803_01 | Female | 57 | A 57-year-old Chinese woman (155 cm, 60 kg, 1.58 m2) presented with lumbosacral/low extremity pain for 2 months. The patient did not have any history of lumbar disk herniation or fracture. Her family history was also negative. Before presenting to our hospital, she had received treatment in several institutions. Initially, she had visited a local hospital and had received acupuncture and moxibustion, which showed no efficiency. A lumbar MRI revealed an abnormal signal in L4, L5, and S1, accompanied by soft tissue shadows in the paravertebral and the spinal canal. Previously, in other hospitals, the patient had received empirical anti-tuberculosis treatment for 4 weeks, which showed poor efficiency. She developed a new symptom of cauda equina syndrome (CES) later. To her relieve the pain and determine the pathology, we performed disk decompression and a biopsy of paralumbar soft tissue. We carried out immunohistochemistry tests to determine the pathology, which indicated that tumor cells were positive for LCA, CD20, CD79, CD10, and BCL-2; were negative for CD3, CD56, MUM-1, CD138, and CD99; and showed a Ki-67 proliferation index of greater than 80%, which is a characteristic of DLBCL. The diagnosis was reconfirmed by the pathologists in our institution. Bone marrow aspiration came out negative and the echocardiogram appeared normal.
Initially, the patient was administered one cycle of the CHOP regimen (cyclophosphamide 750 mg/m2, doxorubicin 50 mg/m2, vincristine 2 mg, day 1, and prednisone 100 mg, days 1-5, administered every 21 days) and six cycles of the R-CHOP regimen (rituximab 375 mg/m2, cyclophosphamide 750 mg/m2, doxorubicin 50 mg/m2, vincristine 2 mg, day 1, and prednisone 100 mg, days 1-5, administered every 21 days). The average relative dose intensity (RDI) was 100%. One month after the last chemotherapy, lumbosacral pain recurred. 18F-FDG PET/CT showed diffuse hypermetabolic foci in C2, C3, and C5 (SUVmax 13.5) and T11-L2 (SUVmax17.8). Considering the response evaluation of the R-CHOP regimen showed progressive disease (PD), local doctors changed one course of the MA regimen (methotrexate 1 g/m2, day 1 and cytarabine 3 g/m2, 12 h apart, days 2-3, administered every 21 days) and two courses of DHAP regimens (dexamethasone 40 mg, days 1-4, cisplatin 100 mg/m2, day 1, and high-dose cytarabine 2 g/m2, 12 h apart, day 2, administered every 21 days), both of which were unsuccessful.
Then, the patient presented to our department. She suffered from severe bone pain and developed paralysis of both lower limbs. Her laboratory tests were normal. A spine MRI revealed an abnormal signal in T8 and L5, and a hip MRI revealed an abnormal signal in the femoral head (Figure 1). We treated her with adjuvant radiotherapy, with a total dose of 30 Gy in 2.0 Gy daily. However, the bone pain lingered. Given that the involved sites in this case were mainly bone lesions and that multi-line chemotherapy including the R-CHOP regimen showed low efficiency, the patient was given repeated infusions of HD-MTX (MTX 3.2 g/m2, iv drip, day 1, administered every 21 days), followed by leucovorin (LV) rescue administration. The patient had an adverse reaction of neutropenic fever after receiving HD-MTX chemotherapy, which improved with symptomatic treatment. After 10 courses of HD-MTX treatment (six cycles of induced chemotherapy and four cycles of maintenance chemotherapy), the patient achieved complete metabolic remission, as confirmed by 18F-FDG PET/CT, and pain lessened. No therapy was applied to the patient after 2 August 2013. The efficacy evaluation showed complete remission during later follow-ups (Figure 2). During the last follow-up (February 2022), the patient showed a high quality of life, was free from pain, and walked smoothly. The treatment process of this case is shown in Figure 3. | case report, methotrexate, primary bone diffuse large b-cell lymphoma, primary bone lymphoma, refractory | Not supported with pagination yet | null |
PMC7475227_01 | Female | 38 | A 38-year-old female at 35-week and 6-day gestation presented at the gyno-obstetrics emergency room of our institute early morning with abdominal pain in the epigastrium. The pain suddenly appeared and aggravated over an hour. Her obstetric history was G 4 P 0+3+0+2, and she has a history of gestational complications. In her first pregnancy, placental abruption occurred at 39 weeks, and the neonate (weight, 4900 g) was born dead after a caesarean operation. The second pregnancy had a diagnosis of severe preeclampsia at 37 weeks, and the neonate (weight, 3800 g) was born after performing a caesarean operation. Finally, in her third pregnancy, she had preterm labour at 36 weeks, and the neonate (weight, 3400 g) was born after a caesarean operation.
During physical examination, the patient was hemodynamically stable, and the bowel sounds were verified during abdominal auscultation. She had an evident general malaise characterized by abdominal pain in the epigastrium associated with bilious vomiting. The clinical evaluation showed active gestation without foetal well-being.
After clinical evaluation, the doctor decided to initiate analgesic and antispasmodic treatment (sodium metamizole, 1 g/2 mL + scopolamine butylbromide, 20 mg/mL + ranitidine 50 mg + hydration with 0.9% NaCl x 1 L). Pain persisted in the patient 1 h after pharmacological treatment despite the administration of analgesics. Foetal heartbeats were 138 per min. Therefore, the patient was referred to the Surgery and General Internal Medicine department.
The Surgery department evaluated the pregnant patient and noted evident pain in the epigastrium and right hypochondrium. They suspected biliary colic, and accordingly, requested auxiliary laboratory tests (Table 1) and imaging studies (abdominal ultrasound) to rule out acute biliary conditions.
The General Internal Medicine department evaluated the patient as she continued to present with severe abdominal pain and suggested optimizing analgesia with the use of opiates. They too suspected biliary colic.
Three hours after admission, the patient continued to experience pain despite the new drug therapy. The abdominal ultrasound report (Table 2) showed probable intestinal involvement associated with foetal bradycardia of 80-90 beats per minute (BPM). Accordingly, the attending gynaecologist decided to admit the patient to the operating room, and obstetric and surgical intervention was performed (Graphic 01).
Obstetric intervention: A median infraumbilical incision was made. Transverse segmental hysterotomy was performed, a female newborn was delivered by a paediatrician. A hysterorrhaphy in 2 planes was made with ligation of both uterine tubes before the surgeons intervened.
Surgical intervention: The surgeons detected necrosis of intestinal loops of the jejunum, ileum, cecum, ascending colon, and proximal 2/3 of the transverse colon (Fig. 1). Surgical resection of the entire necrotized intestinal segment was decided. Side-to-side anastomosis between the duodenum and the transverse colon was performed, and subsequently, the cavity was washed with heated physiological serum. Finally, points were placed at the meso-level of the jejunum to close the anastomotic gap and the wall was sutured (Fig. 1A, B, C).
Postoperatively, the patient was moved to the Intensive Care Unit for normalization of homeostasis, post-sepsis stabilization, and compulsory parenteral nutrition. The patient recovered successfully; a central venous catheter was maintained for nutritional support. The patient was discharged from the hospital and transferred to the Short Intestine Unit of a specialized hospital in Lima, Peru. | cardiovascular diseases, inflammatory bowel diseases, mesenteric ischaemia, pregnant women | Not supported with pagination yet | null |
PMC5576002_01 | Female | 66 | A 66 year old woman, with no significant medical history, presented to her general practitioner with progressive general malaise that had lasted 6 months. She intentionally lost 10 kilograms because she was overweight. For 1 week, she had suffered from fever, night sweats, and fatigue in the lower limbs. She had no history of tuberculosis and, although living in an endemic Q-fever area, she had had no contact with sheep or goats. After 2 weeks she was referred to a rheumatologist who found an increased white cell count of 25.2 x 109/L (normal range 4-11 x 109/L), an increased C-reactive protein (CRP) level of 42 mg/L (normal range <10), and an increased erythrocyte sediment rate of 48 mm/hour (normal range <30 mm/hour). With the differential diagnosis of an unknown autoimmune disease she was treated with prednisone. As the symptoms persisted after 3 weeks, she was admitted to the department of internal medicine at a rural hospital. The initial differential diagnosis included an unknown generalized infection. Therefore, blood cultures were taken, which were negative and remained negative. The blood levels at that time were: an increased CRP of 106 mg/L, an increased white cell count of 20.6 x 109/L, and negative Q fever and lues. A computerized tomography (CT) scan showed a filling defect of the descending aorta and the coeliac trunk (Fig. 1), which was interpreted as a possibly infected thrombosis and was treated with gentamicin and vancomycin. To prevent further growth of a possible thrombus, the patient was treated with therapeutic Nadroparin (Aspen Pharma Trading, Dublin, Ireland). The CT findings suggested positron emission tomography (PET), which showed a hotspot at the filling defect of the descending aorta and a small hotspot in the right tibia, considered to be septic emboli. A magnetic resonance imaging (MRI) scan of the tibia was performed. The differential diagnosis of the lesion in the tibia was inflammation or a malignancy.
The patient was referred to the department of vascular surgery of a tertiary referral hospital. Here the antibiotic was discontinued. On admission, no abdominal abnormalities were found on physical examination. The symptoms of the patient were persistent, but unchanged from the initial presentation. A multidisciplinary treatment team, including a cardiothoracic surgeon, a vascular surgeon, and a microbiologist recommended a magnetic resonance angiogram (MRA). This showed a non-enhancing lesion in the descending aorta without vessel wall involvement, concluding that the filling defect of the descending aorta was less suspicious of malignancy. Seven days after the referral, the patient developed acute abdominal pain. A CT scan was performed, which showed complete occlusion of the celiac trunk as well as infarctions in the left lobe of the liver, spleen, and both kidneys. To prevent further embolization and to limit any ischemia-reperfusion injury, urgent surgical intervention was performed. Because the MRA showed a non-enhancing lesion without vessel wall involvement, the defect was considered to be resectable, therefore endovascular recanalization and stenting was not considered before surgery.
A left thoracophrenic-laparotomy was performed using left heart bypass with a Biomedicus pump (Medtronic inc. Minneapolis, MN, USA). The distal part of the thoraco-abdominal aorta, from the 8th thoracic vertebrae down to the celiac trunk, was resected (Fig. 2A). An embolectomy of the celiac trunk was performed. An interposition graft (Intergard prosthesis o 22 mm, Maquet Getinge group, Intervascular, Athelia, La Ciotat, France) was inserted between the two ends of the aorta with an oblique anastomosis at the celiac trunk (Fig. 2B). The tumor appeared to have spread beyond the vascular wall into some of the intercostal arteries. After 4 days on intensive care, the patient returned to the ward, recovered without complications, and was discharged in good health 10 days after the operation. The total hospital stay was approximately 5 weeks.
Histological examination revealed an epithelioid angiosarcoma. The tumor was present at the proximal resection edge and the embolus was shown to be a malignant tumor of the same kind. The tumor was staged pT4N0M1. Treatment was taken over by an oncologist, who conducted new PET, CT, and MRI scans to determine the course of treatment. In the PET/CT, a few weeks after the surgery, new abnormalities were found in the right femur and os ilium, suspicious of metastasis. The patient underwent palliative treatment. | angiosarcoma, descending aorta, malignancy, primary aortic tumor | Not supported with pagination yet | null |
PMC3934803_01 | Male | 50 | The patient was a 50-year-old man who reported blurred vision and floaters in the right eye that persisted for about 6 months. He then consulted a local ophthalmologist. He was diagnosed with panuveitis and referred to our department. There was no previous ophthalmic history and his general health was good. He had a 10-year history of contact with dogs, but no history of ocular trauma. On presentation, the best-corrected visual acuity (BCVA) in the affected eye was 20/25, and the ocular pressure was normal. Results of slit lamp examination showed inflammatory cells (2+) and infiltration in the anterior chamber of the right eye. The fundus examination showed (1+) vitreous opacities, (2+) vitreous cells, phlebitis, moderate disc swelling, reddening, yellowish-white retinal lesions in the inferotemporal region of the optic disc, and a white exudative lesion near the inferior arcade (fig. 1a). The left eye was normal. Fluorescein angiography showed a hyperfluorescent lesion and dye leakage from the middle to late phases with a hyperfluorescent optic disc. Peripheral blood tests, including blood cell count, erythrocyte sedimentation test, C-reactive protein, and serum angiotensin converting enzyme, were within the normal ranges. Serum toxoplasma antibody and viral antibodies were negative. Chest X-rays were normal. No signs suggested endogenous uveitis, and no clinical findings suggested uveitis due to tuberculosis or syphilitic, fungal, or viral infection. Examination of the patient's serum for antibody to T. canis larvae by ELISA showed an elevated antibody level (fig. 2).
Based on characteristic funduscopic findings, a diagnosis of ocular toxocariasis was made. Systemic albendazole (ALBENZA , GlaxoSmithKline, Brentford, UK) (800 mg twice daily) was prescribed with oral steroids (initial dose 30 mg/day), which resulted in the resolution of the vitreous opacity. However, the lesion near the arcade seemed to be white and enlarged. It was alleviated because the exudative lesion was dried out with treatments after 1 month (fig. 1b). However, 3 months after treatment initiation, the toxocariasis recurred during a treatment-free period (fig. 1c). In addition to the primary lesion, 2 other yellowish-white retinal lesions with vitreous flares appeared. Anthelmintic albendazole (ALBENZA 800 mg twice daily) and oral steroids (initial dose 20 mg/day) were resumed, and the infectious lesions were controlled gradually after another 2 months (fig. 1d). The BCVA in the right eye improved to 20/12.5. Two months after the retreatment was terminated, an intraretinal lesion was observed protruding from the retinal surface into the vitreous cavity (fig. 3a). Examination of this site by HP-OCT confirmed an elevated lesion protruding from the inner retina to the vitreous cavity (fig. 3e). The lesion was in the inner retinal layer, and the retinal pigment epithelium (RPE) and choroid were intact. The anthelminthic medication (800 mg twice daily) was resumed, and the elevated lesion resolved on OCT within the following 8 months (fig. 3b, f). One year after the therapy, signs of inflammation and the elevated retinal lesion had disappeared along with the reduction of blood anti- Toxocara canis antibody titer (fig. 2). Unexpectedly, after 4 months, the elevated lesion reappeared (fig. 3c, g) and anthelmintic monotherapy (800 mg twice daily) was resumed, resulting in lesion control after 2 months (fig. 3d, h). The patient was subsequently treated with anthelminthic drug and steroid tapering. At the last examination after 21 months of follow-up, the BCVA was 20/12.5 with no evidence of active inflammation. The patient continues to be followed untreated without complications. | high penetration, optical coherence tomography, toxocara, uveitis | Not supported with pagination yet | null |
PMC2808574_01 | Male | 45 | A 45-yr-old male had complained of recurrent wheezing and dyspnea for 7 months. He had worked in a herbal manufacturing factory for 8 yr where he classified many kinds of herbal materials, including Wonji, to prepare for processing. He had experienced nasal symptoms including profuse rhinorrhea and sneezing 4 yr after starting work. These symptoms were aggravated when handling Wonji in the manufacturing process, but improved during holidays and vacations. He was a non-smoker, and had no relative with asthmatic symptoms. His serum total IgE level was 663 U/mL using fluorescent enzyme immunoassay, and the total eosinophil count in the blood was 578/microL. A skin prick test with 55 common aeroallergens showed immediate positive responses to Dermatophagoides pteronyssinus, Dermatophagoides farinae, tree pollens, and grass pollens. Methacholine bronchial challenge test revealed 20% decline of FEV1 at the methacholine concentration of 1.9 mg/mL.
Wonji was obtained from the patient's employer and was prepared as previously described. In brief, Wonji was cut into small pieces and extracted into phosphate-buffered saline (PBS; pH 7.5) 1:5 wt/vol, at 4C for 24 hr, followed by centrifugation at 15,000 rpm at 4C for 30 min. The supernatant was dialyzed (the cut-off molecular weight was 6 kDa) against 4L of normal saline at 4C for 48 hr and then used as crude extracts. The protein concentration of the extracts was determined by bicinchoninic acid (BCA) assay according to the manufacturer's instructions (Pierce, Rockford, IL, U.S.A.). The extract was used for the skin prick tests and specific bronchial challenge test.
One mL of normal saline was administered from a DeVilbiss 646 nebulizer (CS & M Instrument Co., Doylestown, PA, U.S.A.) connected to a dosimeter. The subject was asked to breathe the aerosol with tidal breathing, and this was followed by Wonji extract. The forced expiratory volume in one second (FEV1) and forced vital capacity (FVC) were measured using a spirometer (Multispiro-SX , Irvine, CA, U.S.A.) before and 10 min after each inhalation, every 10 min during the first hour, and then every hours for the next 8 hr after the challenge.
The presence of specific IgE antibody to Wonji extract was determined by ELISA, as preciously described. In brief, microtiter plates (NUNC; immunoplate, Roskilde, Denmark) were coated with 50 microL of Wonji extract (100 microg/mL), and then incubated with 50 microL of either the patient's serum, or undiluted sera from five asthmatics who showed negative skin prick test responses to common aeroallergens as well as Wonji extract. After washing, the immunoplate was incubated with 1:1,000 vol/vol biotin-labeled goat anti-human IgE antibody (Sigma, St. Louis, MO, U.S.A.) followed by incubation with 1:1,000 vol/vol streptavidin-peroxidase (Sigma). After washing, 75 microL of TMB solution, 3,3',5,5'-tetramethylbenzidine, one tablet in 10 mL of phosphate citrate buffer containing 2 microL of 30% hydrogen peroxide was added as substrate, and 75 microL of 2.5 N H2SO4 was added to stop the reaction 5 min later. A calorimetric reaction was measured by the absorbency at 450 nm on an ELISA reader. All assays were performed in triplicate.
Competitive ELISA inhibition tests were performed to determine the specificity of IgE binding to a Wonji antigen. In brief, 50 microL of the patient's serum were preincubated with Wonji extract and Der p 2, mugwort extracts for 1 hr at room temperature. The mixture was then incubated on a Wonji-coated microtiter plate for 2 hr. The same steps were then followed as for ELISA. After studying the control samples, in which equal volumes of PBS were preincubated instead of inhibitors, the inhibition of the specific IgE binding was expressed as: 100-(absorbance of samples preincubated with allergens/absorbance of samples preincubated with PBS)x100 (%).
The protein composition pattern and specific IgE binding components of Wonji were analyzed by SDS-PAGE and immunoblot analysis by using the patient's serum. In brief, 25 microg of Wonji extracts were loaded and separated by 12% SDS-PAGE. After electrophoresis, the gel was stained with Coomassie Brilliant Blue R-250 solution (Bio-Rad, Hercules, CA, U.S.A.) and analyzed. To identify the specific IgE binding components of Wonji extracts, immunoblot analysis was performed with the asthmatic patient's serum. After separating the proteins by SDS-PAGE, they were electrophoretically transferred from the gel to a nitrocellulose membrane in a Bio-Rad Trans-Blot system. Blocking was done by incubation in a solution of 10% non-fat dried milk in 0.05% TBS-T buffer, pH 7.5 for 1 hr at room temperature. The nitrocellulose membrane was then washed, cut into strips and separately incubated overnight at 4C, with the patient's serum which had been diluted 1:10 with the blocking solution. The membrane was then washed and incubated with goat anti-human IgE conjugated HRP (Sigma, St. Louis, MO, U.S.A.), in the presence of blocking solution, for 1 hr at room temperature. After further washing, the membrane was incubated in SuperSignalWest Pico chemiluminescent substrate (Pierce, Rockford, IL, U.S.A.) for 5 min. Fluorescence signals were detected by autoradiography using the Kodak Biomax Light ML film (Eastman Kodak Company, Rochester, NY, U.S.A.). | null | Not supported with pagination yet | null |
PMC9048080_01 | Female | 49 | A 49-year-old woman from Bangladesh presented with a three-month history of progressive dysphagia to solids and liquids, dry cough, malaise, and associated 30-pound unintentional weight loss. She was admitted to multiple hospitals due to similar complaints of dysphagia and progressively worsening symptoms, but no formal diagnosis was made. She denied any fevers or hemoptysis. Endoscopic gastroduodenoscopy (EGD) performed in the outpatient setting two weeks prior to admission demonstrated a peptic ulcer but was otherwise unremarkable. She was also diagnosed with right-sided facial nerve palsy a month prior to her admission and was treated with a two-week course of corticosteroids. Her symptoms relapsed after discontinuation of steroid therapy.
The patient's vital signs were stable throughout her admission. On examination, there was significant cachexia and new left facial weakness in a lower motor neuron pattern consistent with facial nerve palsy. She was alert and displayed no signs of cognitive impairment. She was unable to completely close the right eye. There was absence of conjunctival chemosis, proptosis, or periorbital edema. Uveitis was ruled out by an ophthalmologic exam. There was decreased sensation in right V1-V3 distribution, hearing was reduced on right compared to left to finger rub, decreased sensation in the right oropharynx, symmetric rise of the palate, and her tongue appeared midline on protrusion. Cranial nerves were otherwise grossly intact. She had decreased muscle bulk throughout and 5/5 strength in the arms. In the legs, she had bilateral 4/5 proximal strength and 5/5 distal strength. She had normal reflexes in both upper and lower extremities.
The patient's laboratory investigations revealed BUN 3.97 mg/dl and Na 132 mmol/L. She was initially placed on airborne precautions, given constitutional symptoms and positive Quantiferon tuberculin testing, which were subsequently discontinued after obtaining sputum samples that were negative for acid fast bacilli. Chest radiograph demonstrated hilar prominence but no focal airspace opacities (Fig. 1). Computed tomography (CT) of the chest was significant for interval development of extensive bilateral paratracheal, subcarinal, and bilateral hilar lymphadenopathy (Fig. 2) consistent with Garland's triad.
A fiber-optic endoscopy was performed to evaluate dysphagia which revealed immobility of the right vocal fold, significant pooling of secretions, hooding of arytenoids with erythema and edema, and oropharyngeal dysphagia. Initial videofluoroscopic study revealed indentation of the hypopharynx in the region of the cricopharyngeal muscle. The study was aborted given inability to adequately clear moderate puree residue in the pyriform sinuses and proximal esophagus, as well as significant coughing, gagging, and hocking in the absence of aspiration. She was placed on aspiration precautions and enteral feeding was started via nasogastric tube.
Due to bilateral facial nerve palsy, a lumbar puncture was performed which showed an opening pressure 15 mmHg, white cell count of 18 [0-10/UL] with predominant lymphocytes 93% [40-80%], red blood cell of 3, protein 66 [20-100 mg/dl], Glucose 62 [40-170 mg/dl]. Oligoclonal Banding was absent. Various serum and cerebrospinal fluid (CSF) studies were done to evaluate neoplastic, paraneoplastic, infectious, and inflammatory etiologies but were all unremarkable. Serum HIV 1/2 EIA, serum and CSF VDRL, serum HSV 1/2 PCR, serum EBV panel, serum and CSF Lyme disease total Antibody IgG/IgM were negative. Angiotensin Converting Enzyme in serum and CSF were 65 [14-82 U/L] and 2.1 [0.0-2.5 U/L], respectively. Although serum ACE has historically been the most widely known serum biomarker in sarcoidosis, its sensitivity ranges 22-86% and specify 54-95% making their clinical utility a matter of debate.
Magnetic resonance imaging of the brain with and without contrast showed enhancement of the seventh and eight cranial nerves concerning for granulomatous disease or lymphoma (Fig. 3). MRI of the l-spine showed no acute fractures, no compression of the conus or causa equine and no high grade central canal or neural forminal stenosis. Since the CT scan of the chest showed significant lymphadenopathy, she underwent endobronchial ultrasound (EBUS) guided fine needle aspiration (FNA) with multiple lymph node biopsies, which revealed non-caseating granulomas without malignant cells (Figs. 4, Fig. 5). Further testing and interpretation by the was significant for the absence of acid-fast bacilli by AFB stain, absence of fungi by GMS stain, and absence of immunophenotypic evidence of a lymphoproliferative disorder by flow cytometry.
The patient was diagnosed with probable neurosarcoidosis given the clinical presentation, laboratory evidence of CNS inflammation, MRI evidence of granulomatous involvement of cranial nerves VII and VIII, and extra-neural lymph node biopsy suggesting of sarcoidosis with positive histology [Fig. 1] as per the Neurosarcoidosis Consortium Consensus Group. Neural tissue biopsy was not done to confirm the diagnosis in this patient since it is considered an invasive procedure and enough evidence was obtained to initiate treatment without further delay. The patient was immediately started on intravenous methylprednisolone 40mg every 12 h. In light of the need for a long-term course of high dose corticosteroids, treatment for latent tuberculosis and pneumocystis carinii pneumonia prophylaxis were also commenced. She had significant subjective improvement in dysphagia and musculoskeletal symptoms within the first week of treatment. There was modest improvement in the left facial nerve palsy and resolution of lower extremity weakness. Repeat videofluoroscopic study performed one week after steroid therapy showed improved pharyngeal motility. Diet was slowly advanced and intravenous corticosteroid was transitioned to oral glucocorticoid. Follow-ups with rheumatology, infectious disease, neurology, and pulmonology were provided. | null | Not supported with pagination yet | null |
PMC5014938_01 | Female | 65 | We present the case of a 65-year-old (weight, 80 Kg, height, 150 cm) Sri Lankan black woman, known to have LTBI treated with INH and dyslipidemia. She was diagnosed with LTBI based on a positive purified protein derivative test. The patient's main indication for LTBI treatment was recent arrival (within 5 years) from a high prevalence country. She presented to the emergency department with epigastric and right sided flank pain of one-week duration. The history goes back to one week prior to admission when the patient started to experience pain along with one episode of vomiting and dark (tea-colored) urine without any other urinary symptoms. She reported a severe pain of 8/10, relieved by sitting and lying forward and worsened by lying backwards. The patient denied any nausea, diarrhea, constipation, or fever prior to the presentation.
Her past medical history includes LTBI (6 months prior to the presentation with no laboratory and clinical monitoring for the isoniazid treatment) and dyslipidemia without any known liver disease. Her medications are Rimifon (INH) 150 mg two tablets by mouth once daily and Panadol (Acetaminophen) 500 mg two tablets by mouth as needed for headache. The patient was previously on Liponorm (Atorvastatin) 10 mg one tablet by mouth once daily and Neurobion (Vitamin B12, 200 mcg, Vitamin B6, 200 mg, and Vitamin B1, 100 mg) both of which were discontinued when INH was initiated (6 months prior to the presentation). The patient works as a housekeeper; she denied any known drug or food allergies, the use of recreational drugs, tobacco smoke, or alcohol.
Her vital signs were as follows: blood pressure of 120/78 mmHg, heart rate of 83 beats per minute, respiratory rate of 15 breaths per minute, and a body temperature of 36.2 C. Her review of systems was normal except for icteric sclera, pale conjunctiva, and epigastric tenderness. Pending laboratory results, she was given Perfalgan (acetaminophen) 1 g IV STAT for pain and normal saline 0.9% 1 L every 24 hours. The patient's laboratory data upon admission are shown in Table 1.
The patient was admitted to the internal medicine service for further investigation. INH was discontinued and medication orders were the following: Profenid (ketoprofen) 100 mg IV every 8 hours as needed for pain, Nexium (Esomeprazole) 40 mg IV once daily, and Buscopan (Scopolamine Butylbromide) 1 ampoule of 20 mg IV every 8 hours as needed for abdominal pain. Based on negative infectious serology, hepatitis A, hepatitis B, hepatitis C, cytomegalovirus (CMV), and Epstein-Barr virus were all ruled out. The patient had a positive CMV IgG Ab (160 AU/mL) suggesting a previous infection. Hepatic autoimmune disease was ruled out as well based on negative immunology markers. An abdominal ultrasound showed a normal liver size of 11 cm, perihepatic fluid effusion, and pericholecystic edema that are most probably related to hepatitis rather than cholecystitis. CT scan of the abdomen and pelvis showed a diffusely thickened gallbladder wall with neither the evidence of obstructive process nor portal vein thrombosis. After ruling out all the possible causes of acute hepatitis in our patient, the final diagnosis was made as INH-induced acute hepatitis.
Throughout the hospital stay, there was a slight reduction (around 15% from baseline) in liver function tests (LFTs); however INR and bilirubin remained elevated. The patient was clinically improving and her laboratory values were slightly better. Upon physician's order, the patient was discharged on Nexium 40 mg one tablet by mouth daily for one week. The patient did not resume the isoniazid treatment upon discharge after the initial hospital admission.
Six days later, the patient was readmitted to the hospital for abdominal distention and increased upper right quadrant pain that radiated to the flanks and increases with food intake and exertion. She also reported decreased appetite, fatigue, and dyspnea with exertion. Upon readmission, she presented with jaundice, icteric sclera, dark urine, and prominent ascites confirmed by wave fluid test. In the ER, she received Nexium 40 mg IV, Profenid 100 mg IV STAT, and NS 0.9% 500 mL every 24 hours which was increased to 1 L every 24 hours when her blood pressure dropped to 85/55 mmHg. Laboratory values were the following: total bilirubin 27.8 mg/dL, gamma-GTP 95 U/L, ALP 283 U/L, ALT (SGPT) 873 U/L, AST (SGOT) 1582 U/L, amylase 164 U/L, lipase 65 U/L, Cr 0.66 mg/dL, and INR 2.28. Bilirubin was also detected in urine. Accordingly, the patient was admitted to the internal medicine floor for fulminant hepatic failure due to INH.
In an attempt to normalize INR, multiple administrations of Konakion (Phytomenadione) were given. Profenid was discontinued due to the increased risk of bleeding and Buscopan 20 mg IV every 8 hours as needed for abdominal pain was ordered. Furthermore, Primperan (Metoclopramide) 10 mg IV STAT, one vial of Albumin (20%) IV STAT, and morphine sulfate 2 mg SC STAT twice for pain were given. The patient had several episodes of hypotension responsive to fluid resuscitation with NS. The ultrasound showed diffused and clear moderate ascites as well as marked edematous thickening of the gallbladder wall with clear content related to intra-abdominal fluid effusion. The patient developed hypoglycemia (32-37 mg/dL) and oxygen saturation dropped to 86%. Due to deterioration, reduced level of consciousness, and poor response to oxygen, she was intubated and transferred to the intensive care unit.
On physical exam she was unresponsive, her pupils were middilated, and nonreactive, her lungs had few scattered rhonchi, and she had abdominal distention and ascites. She had metabolic acidosis (pH 6.8) and multiple doses of sodium bicarbonate were administered. The patient was consistently hypoglycemic and hypotensive despite fluid resuscitation and vasopressor administration. Serum creatinine increased from 0.66 mg/dL to 2.34 mg/dL in two days confirming acute kidney injury. She was also suffering from severe coagulopathy and hepatic encephalopathy. Additionally, blood cultures revealed a sensitive Escherichia coli and sputum cultures showed alpha-Streptococcus and Neisseria species. The patient had cardiorespiratory failure necessitating cardiopulmonary resuscitation (CPR). Spontaneous circulation was achieved after 15 minutes of CPR. Unfortunately, this was followed by a second cardiac arrest and despite maximal hemodynamic support, the patient passed away. | null | Not supported with pagination yet | null |
PMC5529052_01 | Male | 70 | A 70-year-old man presented to the Jefferson Pancreas, Biliary, and Related Cancer Center for an opinion regarding surgical intervention for a pancreatic mass discovered 1 year earlier, during workup for epigastric pain, thought to be heartburn. He had no history of pancreatitis or obstructive jaundice. He also had no weight loss or steatorrhea. The patient did, however, develop new onset diabetes one year ago.
Routine hematology and metabolic studies were normal. The tumor marker CA19-9 was slightly elevated at 41 U/mL (<35 U/mL) and the CEA was normal at 2.1 ng/mL (<4.7 ng/mL). An abdominal MRI with and without contrast revealed pancreas divisum with a dominant 2.4 cm multicystic uncinate process lesion communicating with the main pancreatic duct (duct of Wirsung) with an associated uncinate duct dilation to between 4 and 5 mm (Fig. 1). Imaging also showed simple hepatic cysts and minimal steatosis, but no evidence of metastasis. The lesion was believed to be an IPMN, based on this study and previous CT scans.
The patient underwent an open cholecystectomy and PPPD without complication. There was no evidence of malignant ascites, carcinomatosis, omental implants, or metastatic disease. Pathological study of the surgical specimen revealed the lesion to be an IPMN with intermediate-grade dysplasia without any invasive component. All surgical resection margins and 15 specimen lymph nodes were negative for malignancy.
The patient was placed on our institution's Whipple Accelerated Recovery Pathway (WARP) with the goal to discharge on postoperative day 5. However, he did develop a slight ileus, which prolonged his stay by 1 day. He was subsequently discharged on postoperative day 6. The patient and his family were appropriately educated on the postoperative recovery expectations, exercise recommendations, diet, and medications. He has fully recovered from his pancreatic resection and is scheduled to have annual surveillance of his pancreatic remnant through MRI/MRCP. | intraductal papillary mucinous neoplasms, pancreas divisum, uncinate process | Not supported with pagination yet | null |
PMC6560993_01 | Male | 50 | A 50-year-old Asian male was referred to our outpatient department from a local clinic with a diagnosis of familial benign pemphigus for 10 years and well controlled hypertension for 20 years now. With the current hospital visit, he had been complaining of waxing and waning lesions under armpit and groin which where dark-reddish in colour with many papular-vesicles which could easily break and leave erosions and ulcers in these regions and later on form scab. They were associated with lots of pain which was initially itchy and fever for the past five days' prior presentation to our hospital. This presentation limited his normal daily chaos. He has not received much improvement from the usual topical and oral medication which he has been taking from since past the illness started.
The review of other system was unremarkable. He does not have diabetes mellitus, endocrinological nor any autoimmune disorder. He is retroviral status non-reactive and neither does he have any infectious disease like tuberculosis, syphilis, hepatitis A, B, C. He has a positive family his of similar clinical presentation in the late mother. He was febrile to touch with axillary temperature of 38.0 C and other vitals where within normal range. Dermatological exam reviewed, diffuse edematous dark erythematous lesions seen in both axillaries and groins as shown in Figure 1 and Figure 2. Erythematous lesions had associated erosions, crusts, blisters, pigmentation, and scale at friction sites, accompanied by pain, itching and malodor. Erosive surfaces left by the ulceration of blisters to be seen with a small amount of exudation and scab on the surface. Nikolsky's sign was positive.
The rest of the examination was unremarkable. He was treated as Hailey-Hailey disease in view of the positive family history in mother, characteristic lesions in suspect flexure areas, relapsing course, nature of the illness and the referring clinicians' diagnosis. However, he did not show the classical asymptomatic linear, white bands in the nail plates as documented by many authors. We did not do a biopsy and PCR for genetic mutation of ATP2C1 to confirm diagnosis due to financial constraints. All radiologic investigations were normal.
The patient was empirically initiated on both topical and systemic agents for disease modifying and symptomatic treatment: injectable glycyrrhizin acid, analgesic- codeine phosphate, anti-histamine; cetirizine for symptomatic relief and topical cefoperazone tazobactam in view of the fevers. Day 3 on this cocktail showed little improvement in disease improvement with blood test results suggesting that the disease is in active stage thus, an immunomodulator, azathioprine orally was added to the regimen. Other supporting drugs where directed by laboratory findings. For hypoalbuminemia, he was advised on a high-quality protein diet and fluid ingestion in view of the high uric levels.
Most supportive investigations done where unremarkable as indicated in Table 1. Of note was the raised markers of inflammation, erythrocyte sedimentation rate of 64mm/hour, positive skin exudation culture for Streptococcus agalactis, monocytosis of 15.6% monocyte absolute value: 1.15 x 109/L. He also had hypokalemia and hypoalbuminemia and hypoglobuminemia. Laboratory biochemistry tests, complete blood count, serum viral screen, autoimmune screen; ANA+ENA, ANCA spectrum, GM antibody determination and rheumatoid factor, TORCH screen infections, potassium hydroxide scrapping for fungal, fungi 1-3-B-D dextran quantitative G test, Stool and urine analysis were unremarkable.
The patient showed much improvement after day 5 on azathioprine and he was treated as outpatient on the same cocktail with a 2 weekly supply of the immunomodulator as indicated in Figure 3, Figure 4. | familial benign chronic pemphigus, hailey-hailey disease, azathioprine, benign chronic familial pemphigus, benign chronic pemphigus, familial benign pemphigus | Not supported with pagination yet | null |
PMC7227740_01 | Male | 42 | A 42-year-old man presented with the compliant of cough and breathlessness (modified medical research council grade-II) for the last 5 years with mild non-documented fever (on and off) for last 6 months. There is no significant past history. He was on antitubercular therapy (ATT) for last 3 months on basis of clinic-radiological diagnosis with no improvement in symptoms. He is a scissor/knife sharper by occupation with exposure for iron dust for the last 25 years. He is a non-smoker. On examination, general physical examination was normal. On respiratory examination, there was bilateral vesicular breath sound with bilateral fine crepitation. Routine haematological and biochemical investigation were normal. X ray of chest (postero-anterior view) showed bilateral nodular opacities in all zones [Figure 1]. Sputum for acid fast bacilli (AFB) direct smear was twice negative. Connective tissue serological profile (ANA, Rheumatoid factor, p-ANCA/c-ANCA, Scl-70) was negative. Serum angiotensin converting enzyme level was 118 U/l. His 6-min walk test (6MWT) was 320 m with desaturation of 9%. Pulmonary function test (PFT) showed FVC-2.63lts (73%), FEV1-1.31lts (44%), FEV1/FVC-50, TLC-5.06 (99%) and DLCO-23.56 (88%). HRCT chest showed numerous reticulonodular densities in bilateral lung fields and multiple nodular lesions in the sub-pleural aspect of the right lower lobe with fibrolinear densities in both upper lobes [Figure 2a-d]. Bronchoscopy showed normal bronchial tree. Bronchoaveolar lavage (BAL) and Transbronchial lung biopsy (TBLB) were obtained from the right middle lobe. The AFB smear and geneXpert of BAL fluid were negative. The bacterial and fungal cultures from the BAL fluid were sterile. TBLB showed heavy deposition of dark to brown pigment in perivascular and peribrochiolar areas with mild chronic inflammation and focal interstitial fibrosis. Iron stain shows fine and coarse granules in macrophages on prussian blue. [Figure 3a and b] He was finally diagnosed as pulmonary siderosis, ATT was stop and advised to change occupation and use of protective gear while working. | occupation, sharpening, siderosis | Not supported with pagination yet | null |
PMC9905019_01 | Female | 58 | A 58-year-old woman, originally from Somalia, presented to our Dermatology clinic for assessment of a relapsing course of generalized pruritic eruption. Her comorbidities included type 2 diabetes, hypertension, dyslipidemia, non-alcoholic fatty liver disease, peripheral artery disease, and immune thrombocytopenic purpura. She reported that her skin lesions improved with sun exposure and worsened in the winter. Previous treatments included only calcipotriol and betamethasone dipropionate ointment to which she had minimal improvement. On physical examination, there were widespread scaly erythematous papules and plaques, affecting 10% of her body surface area (BSA). Halobetasol propionate cream was started while awaiting histopathology results that finally confirmed a diagnosis of psoriasis.
As a treatment for her severe psoriasis (baseline BSA 10%, Psoriasis Area Severity Index (PASI) of 10.2; Dermatology Life Quality Index (DLQI) of 15) and patient preference against a biologic therapy at this time, apremilast 30 mg twice daily was initiated. After 16 weeks of treatment, her psoriasis significantly improved (BSA of 2%, PASI of 2.6). However, after 4 months of treatment, the dosing had to be reduced to 30 mg daily due to gastrointestinal side effects. Following this dose modification, her psoriasis worsened (BSA of 15%; PASI of 12.8), and to prepare her for the next therapeutic option, we performed a pre-immunosuppressive work-up that included blood test, TB skin test, and chest X-ray (CXR).
At this pre-biologic screening, she was diagnosed with LTB by the infectious diseases team. She had an abnormal CXR, and positive TB skin test and QuantiFERON-TB Gold without any symptoms of active TB. A computed tomography (CT) chest scan was ordered for further assessment, showing a small region of chronic inflammatory reaction in the posterior area of the left upper lobe and a superimposed granuloma. She was treated with rifampicin 600 mg daily for 4 months, and much to our surprise, her psoriasis completely resolved within 2-3 months of treatment. In addition, her psoriasis has remained clear 1 year after stopping rifampicin. | psoriasis, biologics, infection, rifampicin, tuberculosis | Not supported with pagination yet | null |
PMC9659588_01 | Female | 15 | A 15-year-old Chinese girl suffering from fatigue, dizziness, and nausea was presented with severe bone marrow pancytopenia in October 2019. Acute jaundice hepatitis of unknown cause had been diagnosed 1 month before admission to our hospital. Complete blood count (CBC) showed that white blood cell counts were 0.19 x 109/L, absolute neutrophil count (ANC) was 0.11 x 109/L, hemoglobin level was 76 g/L, and platelet count was 12 x 109/L. Blood exams showed that the transaminase and bilirubin were 20 times higher than the normal range (total bilirubin (TBIL) 251.7 mumol/L, direct bilirubin (DBIL) 179.3 mumol/L, indirect bilirubin (IBIL) 72.4 mumol/L, aspartate aminotransferase (AST) 1420 IU/L, alanine aminotransferase (ALT) 1757 IU/L). T-cell subsets of peripheral blood detected by flow cytometry showed that CD4+ was 9.11%, CD8+ was 69.83%, and CD4/CD8 rate was 0.14, which the ratio of CD4+/CD8+ reduced significantly. Bone marrow biopsy showed extremely hypocellular bone marrow (5%), reduced granulocytes and erythrocytes, hyperplasia of adipose components, no megakaryocytes, and an increased number of lymphocytes, plasma cells, and histiocytes. A cytogenetic study showed a normal karyotype. Serological tests for hepatitis (A, B, C), Epstein-Barr virus, cytomegalovirus (CMV), parvovirus B19, herpes, and HIV were negative. No positive genetic and molecular tests were found for congenital bone marrow failure diseases such as Fanconi anemia, Shwachman-Diamond, and dyskeratosis congenita. Paroxysmal nocturnal hemoglobinuria (PNH) diagnosis also was excluded. HAAA with negative viral serology was finally diagnosed.
Treatments with liver protection, nutrition support, and immunosuppressive therapy (IST) with prednisone, cyclosporine (CsA), and hematopoiesis stimulation (granulocyte colony-stimulating factor (G-CSF), thrombopoietin receptor agonist (TPORA), and danazol) were started after admission. At the beginning of hospitalization, the patient refused antithymocyte globulin (ATG) or HSCT treatment due to the family's financial distress. Due to pulmonary infection from agranulocytosis, beta-lactamase inhibitors tazobactam (piperacillin-tazobactam, cefoperazone-sulbactam) and fluoroquinolones (levofloxacin) were used, successively. The patient's liver function improved, and transaminases were back to normal after 1 month of liver protection treatment. However, the pancytopenia treatment showed no significant response. The patient remained platelet and red blood cell transfusion-dependent. Without effective infection control, the patient developed an oral fungal infection (Figure 1A) and pulmonary infections with MDR to bacteria and fungus due to long-term neutropenia and antibiotic use. An aspergilloma and a large number of pleural effusions were found by CT scanning (Figure 2A). Laboratory tests repeatedly showed sputum culture with Pseudomonas aeruginosa resistance to multidrug in December 2019. Serum (1,3)-beta-D glucan (G-test) was negative, while galactomannan (GM-test) was positive. The bronchoalveolar lavage was rejected by the patient, and multiple fungal cultures from the sputum showed negative results. A conventional microdilution assay (CLSI methodology) showed the minimum inhibitory concentration values of imipenem and meropenem of 8 and >16, respectively. Empirical antibiotics Fosfomycin, Polymyxin, Meropenem, Voriconazole, and Micafungin could not control the patient's infections. The patient's symptom of fever, cough, and shortness of breath continued. Oral ulceration with fungal infection aggravated, and multidrug-resistant infection progressed to life-threatening sepsis. At the same time, the patient presented with vomiting blood and developed hemorrhagic shock. After effective antishock treatment, the patient's condition was under control. With uncontrolled infections after multiple antibiotic treatments, the patient was given Ceftazidime Avibactam Sodium (2.5 g/kg i.v. q8h) and Fosfomycin (4 g b.i.d., i.v.) with a continuous infusion of Voriconazole and liposomal amphotericin B. At the same time, continuous drainage of the pleural fluid was performed. The patient's status gradually improved, although the size of the fungus ball in the right upper lung showed no obvious shrinkage. The level of inflammatory markers of C-reactive protein (CRP) decreased from 175 to 68 mg/L, procalcitonin (PCT) from 3.5 to 0.1 ng/ml, and IL-6 from 1200 to 126 pg/ml (Figure 3). CT scan showed the bilateral pleural effusion was reduced significantly. The patient recovered from the hemorrhagic shock from gastrointestinal bleeding.
As the patient's health improved, we urgently considered hematopoietic stem cell transplantation with salvage therapy. When the patient family's financial situation improved, peripheral blood stem cells from her HLA-matched sister were transplanted. The conditioning regimen of peripheral blood stem cell transplantation was 2.5 mg/kg rabbit ATG (on day -5 to day -2) and 50 mg/kg/day cyclophosphamide (on days -5 to -2) from December 25 to 29 2019. Peripheral blood stem cells (CD34+ 10.15 x 106/kg) were infused on day 0 (December 302019). CsA with the goal serum level in the range of 200-300 ng/ml was started on day -1. In total, 15 mg/m2/day MTX (day +1), 10 mg/m2/day MTX (+3, +6), Mycophenolate (day +7 to +45), and ruxolitinib (5 mg, bid) were administrated for graft versus host disease (GVHD) prophylaxis after neutrophil engraftment. MTX on day +11 was discontinued to reduce the risk of hematological toxicity. On day +15 post-transplantation, the patient's ANC was 0.5 x 109/L and it continued to increase. G-CSF was discontinued 3 days after ANC reached >1 x 109/L. Platelet engraftment occurred on day +35. Then, the antibiotics were switched to antibacterial (cefoperazone/sulbactam, linezolid) and antifungal (caspofungin, liposomal amphotericin B) therapies. The patient's temperature was returned to normal since day +18. The CT scan showed aspergilloma in the right upper lung with bilateral pleural effusion and left atelectasis on day +22, which seemed more serious than before HSCT. That might be because the patient suffered prolonged neutropenia before granulocyte engraftment (Figure 2B). With the patient's health conditions improving rapidly, atelectasis and bilateral pleural effusion gradually disappeared. Shrank aspergilloma was seen on day +59 (Figure 2C). The patient's CBC returned to a nearly normal range (WBC of 6.5 x 109/L, Hb of 100 g/L, and platelets (PLT) of 104 x 109/L) on day +72 with normal liver and kidney function (May 1 2020). Oral fungal infection was cured (Figure 1C).
After allo-HSCT, the patient's condition continued to keep stable. Donor chimerism of 100% was achieved. Slightly chronic skin GVHD was observed and was well controlled with methylprednisolone. Following-up 26 months after transplantation, the patient was still in complete remission with a high-quality life. She was back to school after 1 year of allo-HSC transplantation. | hematopoietic stem cell transplantation, hepatitis-associated aplastic anemia, multidrug-resistant bacteria, severe infection, very severe anemia plastic | Not supported with pagination yet | null |
PMC6104492_01 | Female | 26 | Alice (a pseudonym) was a single-26 year old employed woman who was treated for depression at the Maia University Institute (ISMAI), Portugal. She participated in a randomized clinical trial called the ISMAI Depression Study, which compared the efficacy of Cognitive behavioral therapy (CBT) and Emotion Focused Therapy (EFT) for patients diagnosed with major depressive disorder.
Psychological treatment, as well as the collection and processing of data for research purposes followed principles and standards included in the ethics code (American Psychological Association's - APA - Ethical Principles of Psychologists and Code of Conduct, as well as the Code of Ethics of Portuguese Psychologists). The client of this study, like all other participants, signed an informed consent required in the Standard 3.10 of the ethics code. Previously, the client was informed about the purposes of the research, expected duration and procedures. In addition, it was clarified that participation was voluntary, preserving their right to refuse participation or to give up participating at any time. In this informed consent, Alice also authorized the use of the collected data for process and outcome studies.
Alice received 16 weekly sessions of EFT and was considered a good-outcome client, as described later. She lived with her parents, who were catholic and conservative. Her presenting problems concerned her relationships with her boyfriend and with her parents (mainly her father) and problems at work. Mendes and collaborators previously classified categories of setbacks in this case.
Alice's therapist was a Portuguese woman in her early thirties with 8 years of experience as a therapist, including 4 years of experience delivering EFT.
Emotion Focused Therapy is an empirically validated humanistic therapy that views emotions as an essential element in human functioning. According to this therapeutic model, emotions signal important needs underlying people's experiences and promote adaptive action tendencies, helping them adapt and survive. Psychological problems are viewed as consequence of maladaptive emotional processing. The main therapeutic goal is to change this maladaptive emotional processing, allowing adaptive emotions to emerge and promote more adaptive functioning.
The Portuguese version of the BDI-II (translated into Portuguese from) is a 21-item self-report questionnaire that assesses depressive symptoms. Higher total scores indicate severe depressive symptoms. For the Portuguese population, significant clinical depressive symptoms are signaled by a total score higher than 13. The results of the Portuguese validation were considered good. Internal consistency reliability measured by Cronbach's Alpha was 0.89.
The OQ-10 is a self-report questionnaire composed by 10 items that measures health functionality. Each item is scored on a scale ranging from 0 to 4 and the total score goes from 0 to 40. Obtaining a higher score in this questionnaire indicates the presence of poorer mental health functionality. The OQ-10 Cronbach's Alpha was of 0.88 (Seelert, unpublished) and the test-retest reliability of 0.62. In the sample from ISMAI Depression Study (n = 64), the internal consistency was of 0.88 (Cronbach's Alpha) and the test-retest reliability was of 0.74 over a 1-week interval.
As summarized in Table 1, the APES describes the evolution of the relation of a problematic experience (or voice) to the self (dominant community of voices) using a sequence of eight stages, numbered 0 to 7, ranging from warded off (i.e., muted or dissociated) to mastery (i.e., fully integrated and no longer a problem, serving as a resource in new situations). Theoretically, the APES is considered as a continuum, and intermediate ratings (e.g., 2.3, 4.6) are allowed. However, for this study, we used only whole numbers on the APES (e.g., 2, 3).
The BDI-II was administered at initial and post-treatment assessments and at sessions 1, 4, 8, 12. The OQ-10 was administered immediately before each session. Alice's 16 sessions were videorecorded and later transcribed verbatim following the transcription conventions described by.
Alice met criteria for the ISMAI Depression Study, which included: being diagnosed with Major Depression Disorder; Global Assessment of Functioning > 50. The exclusion criteria were: currently on medication or another form of treatment; or currently or previously diagnosed with one of the following DSM-IV Axis I disorders: panic, substance abuse, psychotic, bipolar, or eating disorder; or one of the following DSM-IV Axis II disorders: borderline, antisocial, narcissistic, or schizotypal; or at high risk of suicide. Screening for inclusion and exclusion criteria used the Structural Clinical Interview for the DSM-IV-TR. After being admitted to the study, each client was randomly attributed to a therapeutic condition (CBT or EFT) and, afterward, randomly assign to a therapist.
Alice's scores on the BDI-II the ISMAI trial's criterion measure, declined from 29 at initial assessment to 1 at her last session and 5 at 1-year follow-up. She was considered a good-outcome case because she met criteria for clinically significant and reliable improvement, as described by: (a) Her scores improved from above to below the cut-off of 13 on the Portuguese BDI-II, indicating clinically significant improvement, and (b) the amount of change was greater than the reliable change index of 7.75, that is, a difference greater than likely to have occurred by chance (at p < 0.05), indicating reliable improvement. For this case study, she was selected from among the ISMAI trial clients who met the criteria on the basis that complete transcripts were available.
Our assimilation analysis followed procedures used in previous studies (e.g.,).
The two APES raters were a PhD clinical psychologist, and a Ph.D. student in clinical Psychology, both with previous experience in research on the assimilation model. Training took approximately 4 months and included weekly meetings in which journal articles about the assimilation model were read and discussed and, sample sessions were coded according to the APES until all raters were considered reliable, achieving an intraclass correlation coefficient reliability of ICC [2,1] >= 0.60.
Next, both members of the team read transcripts of the entire case and identified the main recurring issues. By consensual agreement, they chose and characterized two main themes based on clinical relevance and time spent in therapy. The first theme was "fear of being rejected and abandoned," which concerned Alice's difficulty in imposing her needs to others, motived by an intense fear of not being accepted for what she was. The other theme selected was "hurt toward her father," which concerned unfinished business with her father concerning an episode in which, at the age of 15, she discovered an affair her father was having. She had never confronted him about this, but she resented his infidelity. Alice's dominant voice was labeled as "fear of being rejected," as she presented a similar interpersonal pattern across contexts: work, relationships with family and boyfriend. The problematic voice for both themes was labeled as "I have the right to express myself and be accepted," characterizing similar experiences of wanting to assert her needs and rights. These two voices seemed closely similar for both of these themes.
After selecting the themes, raters excerpted all passages where the themes appeared (N = 554) and rated them, independently according to the APES. The passage was the rating unit and was defined as a stretch of discourse delineated by a change in the topic of the conversation or by markers of changes in APES level (see). Reliability of these independent APES ratings was assessed using the Intraclass Correlation Coefficient designated ICC (2,2) by, which is the reliability of the average of two raters. The ICC (2,2) was of 0.966, which is considered good. Subsequently, raters discussed and reached consensus on APES ratings of passages where they disagreed (see). The consensus ratings were used in our analyses.
Figures 1, 2 show the assimilation progress in each theme, across passages. Passages dealing with particular themes were not evenly spaced across sessions, and the divisions shown in the figures are meant to indicate the approximate parts of the treatment in which each theme was addressed. The "fear of being rejected and abandoned" theme was much more frequent than the "hurt toward her father" theme. However, because both themes involved the same problematic and dominant voices, suggesting they were different expressions of a common core problem, we decided to combine these themes for our analysis. We also noted that the evolution of the two themes, considered separately, appeared very similar in terms of both APES levels and instability. Thus, to obtain mean APES levels for each session, we averaged APES ratings across passages of both themes within each session.
Assimilation of problematic experiences scales fluctuation in each session was indexed using a measure fully described by, which assessed the amplitude and the frequency of changes in APES levels across one session. In effect, the APES fluctuations index is a way to index the incidence of APES setbacks. Fluctuation values can vary from 0 (low fluctuation) to 1 (high fluctuation). That is, the formula yields a normalized fluctuation intensity, 0 <=F <= 1:
where
yi = |xnk + 1 - xnkj
xn = nth session score
k = points of return (changes in slope in the data sequence)
i = periods between points of return
I = total number of such periods within the window
m = number of measurement points within a moving window
m - 1 = number of intervals between all measurement points of a window
s = xmax - xmin with xmin smallest value of the scale, xmax largest value of the scale.
In the present study we employed a dynamic factor model that is a vector-autoregressive (VAR) method to measure contemporaneous correlations and time-lagged regressions in multivariate time series, using a structural equation model framework. The first step for this analysis was to smooth the time-series data and remove its trend. A moving average was calculated for the APES level, APES fluctuation, and symptom intensity measured by the OQ-10. The moving average was calculated by taking the arithmetic mean of a moving-window of four sessions. Total observations within each variable was equal to 16, the total number of sessions of Alice's treatment. For the dynamic factor analysis, we treated each variable as a separated dependent variable.
A VAR model with a lag of 1 session was tested by creating a block-Toeplitz matrix. The VAR model allowed to analyze intra- and intersession relationships between each variable. All VAR analyses were carried out in LISREL (Version 8.80). Since we had a single indicator for each latent variable, the lambda (lambda) was set to identify and the beta (theta) matrix was fixed at zero. The beta matrix was then analyzed to search for meaningful modification indices (MIs). To improve our model, whenever there was a significant MI, we rerun the analysis with the significant path. The final model was accepted when no more significant MIs were observed within the matrix. | assimilation model, depression, dynamic factor model, dynamic systems theory, fluctuation, instability, psychotherapeutic change process | Not supported with pagination yet | null |
PMC9396231_01 | Female | 54 | A 54-year-old woman was referred to our hospital for assessment of 'multiple round cystic opacities varying in size' seen on a chest radiograph (Fig. 1A and B) and a concern for possible lung malignancy. She had been a homemaker for many years in a rural area of Eastern Cape Province, with extensive exposure to indoor biomass fuel, and had close contact with dogs, cattle and sheep. She presented with a 6-month history of daily cough, productive of half a cup of yellow/green sputum that had become white over the past month. She had noted haemoptysis on only two occasions during the first month of cough. She described seeing white 'eggshell membrane'-like substances in her sputum over several months. During the same period, she experienced shortness of breath on exertion, wheeze that was worse at night, central sharp chest pain that radiated to both arms, and left upper-quadrant (LUQ) that was worse when lying down on her left side. Constitutional symptoms included weight loss (15 kg), with no associated loss of appetite, and night sweats that were profuse at the start of the symptoms, with associated fever and chills. These symptoms subsided somewhat after two courses of antibiotics.
She was a non-smoker and had no history of tuberculosis (TB); previous investigations for TB were negative (GeneXpert (Cepheid, USA) and auramine stain). She recalled being diagnosed by a local doctor, with two abdominal masses on ultrasound examination, but defaulted on her appointment at a tertiary centre to which she had been referred.
On physical examination, she had a blood pressure of 118/80 mmHg, heart rate of 80 bpm and respiratory rate of 22 breaths per minute, with saturation of 94% in room air. She was comfortable and weighed 50.0 kg (body mass index (BMI) 20.8). A general examination was normal, and her respiratory examination revealed a trachea deviated to the right and dullness in the right upper and lower zones, with decreased breath sounds in the right lower zone. She had bronchial breathing globally with no wheeze or crackles. Her abdomen was soft and non-tender, with no ascites. She did have mild hepatomegaly and an 8 - 10 cm firm mass in the LUQ.
On further investigation, her enzyme-linked immunosorbent assay (ELISA) HIV test was negative and a full blood count revealed a mild microcytic anaemia (Hb 10.7 g/dL, mean corpuscular volume 78.3 fL, mean corpuscular Hb concentration 25.2 pg, white cell count 7 230/muL, platelet count 5 810/muL) and elevated lactate dehydrogenase (248 U/L). She had normal liver enzymes, international normalised ratio (INR) and renal function (total protein 85 g/L, albumin 35 g/L, total bilirubin 4 mumol/L, conjugated bilirubin 2 mumol/L, aspartate transaminase 20 U/L, alanine transaminase 15 U/L, alkaline phosphatase 95 U/L, gamma-glutamyl transferase 19 U/L, INR 1.30, urea 3.7 mmol/L, creatinine 76 mumol/L).
Sputum-microscopy sensitivity and culture revealed +3 neutrophils with normal respiratory flora. GeneXpert was negative for Mycobacterium tuberculous. Pulmonary function tests demonstrated mild obstruction with significant and complete reversibility after administration of a bronchodilator (260 mL; 19%) and normal diffusion capacity (forced expiratory volume (FEV1 ) 1.37 L (63% predicted), forced vital capacity (FVC) 2.02 L (79%), FEV1 /FVC 67.69%, and carbon monoxide transfer factor 16.42 mL CO/min/mmHg (75% predicted)).
The chest radiograph (Fig. 1A) showed right upper-lobe volume loss with the trachea deviated to the right, crowding of ribs, right upper-zone patchy opacity with possible intracavity mass with crescent of air and marked pleural thickening, large posterior right lower-lobe round opacity (Fig. 1B) and smaller right middle-lobe round opacity with marked bronchial wall thickening but no pleural effusions. The most likely diagnosis was hydatid disease in an endemic area. Other diagnostic considerations included lung cancer, sarcoma or aspergilloma, for which a computed tomography (CT) scan of the chest was done as a further investigation. A CT scan of the chest (Fig. 2A) revealed a large right lower-lobe cyst (8 x 8 cm), with significant right middle- and upper-lobe scarring, traction bronchiectasis/bronchiolectasis and lung cavities containing low-density material with gas locules, in keeping with aspergillomas. The left lung had a small well-circumscribed superior lingual cyst (19 mm), while the upper abdomen (Fig. 2B) revealed two large cysts in the liver (9.5 x 7.0 cm) and spleen (15 x 10 cm). The final diagnosis was hydatid disease of the lungs, with a rare giant splenic hydatid, while the right upper-and middle-lobe disease was possibly due to post-TB lung disease with aspergillomas and/or bronchiectasis. The thoracic surgeons recommended surgical removal of the cysts, which the patient declined, citing a need to consult further with her family. A trial of medical treatment (albendazole 400 mg twice daily) was commenced while awaiting surgery. The patient later became loss to follow-up. | echinococcus granulosus, south africa, hydatid cyst, multi-organ hydatid disease | Not supported with pagination yet | null |
PMC7796834_01 | Female | 28 | A 28 years old female patient, with a six months history of a chronic myeloid leukaemia (CML) treated by tyrosine kinase inhibitors leading to clinical and biological improvement, was admitted in the hematology ward on the 28th of June, due to a 4 days history of diarrhea and fever up to 40 C. White blood cell count showed a leukocytosis of 100000 el/mm3, with a blast percentage of 90%. A bone marrow aspiration and immunophenotyping were realized and revealed a blastic transformation of her CML into an acute myeloid leukaemia (AML) M4 subtype. A chest X-ray was performed as part of the septic workup showing a right lower lobe pneumonia, followed by a chest CT scan (Figure 1) showing bilateral condensation and ground glass opacities in favor of an infectious pneumonia associated with a small amount of fluid in the right pleural cavity. The patient received antibiotics and a first cure of chemotherapy.
As routine evaluation in all patients with hematological malignancies, the patient was tested for SARS-CoV-2. Reverse transcriptase polymerase chain reaction (RT-PCR) from nasal and pharyngeal swab came back positive. Following a deterioration of her general health status, the patient was transferred to the ICU on the 2nd of July for SARS-CoV-2 pneumonia. Upon admission, the medical examination revealed a conscious patient (GCS 15/15), respiratory rate at 46 cycle per minute, oxygen saturation (SpO2) of 70% at room air and 95% upon wearing a non-rebreather face mask (15 L/min), blood pressure of 100/50 mmHg, pulse of 110 bpm and a temperature of 40 C. Complete blood count showed a leukocytosis, white blood cells of 158140 el/mm3, neutrophils of 1580 el/mm3, lymphocyte of 6330 el/mm3, anemia with a hemoglobin at 6.2 g/dl, thrombocytopenia with platelets of 32000. Blood hemostasis analysis showed a biological disseminated intravascular coagulation (DIC), prothrombin time at 48% and partial thromboplastin time at 39.7 sec, fibrinogen at 2.58, D-dimers at 6700 mg/l. Biochemical profile showed an hypokalemia of 1.5 mmol/l, CRP of 106, PCT of 1.68, ferritin at 2000 ng/l and troponins at 70 ng/l, without any tumour lysis syndrome was absent: uric acid< 5, LDH at 3271 UI/l, potassium at 1.5 mmol/l, phosphorus at 27 mg/l, calcium at 85 mg/l. A pretherapeutic EKG showed a sinus tachycardia at 100 bpm, no repolarization trouble and a normal QTc at 464 ms. The echocardiography showed: for the left ventricle: normal in size with normal wall thickness and systolic function, an ejection fraction at 50%, without neither dyskinesia nor thrombosis. For the right ventricle: no valvular leakage or stenosis, a right ventricle systolic function associated with a concentric left ventricular hypertrophy. Both aorta and the inferior vena cava are normal.
An infectious workup, consisting of urine cytobacteriological examination, stool tests, a PCR based detection of mycobacterium tuberculosis, blood cultures and aspergillus serology all came back negative. Therapeutic management included oxygen therapy, non-invasive ventilation at 50% FIO2 (PEP at 5 and IA at 12). Medical treatment associated hydroxychloroquine 200 mg 3 times a day, azythromycin 500mg the first day then 250mg per day, methylprednisone at 80mg a day for 7 days and curative anticoagulation treatment including enoxaparin 100 UI/kg (1mg/kg) twice a day. On day 3, the patient was eupneic (SpO2of 98%) wearing nasal cannula at 3L/min, she no longer needed sessions of non-invasive ventilation. One week later, the patient had a favorable clinical and radiological (Figure 2) outcome and was declared cured of the SARS-CoV-2 infection following a negative PCR test, she was transferred back to the hematology ward for further treatment of her acute myeloid leukaemia. | covid-19, leukaemia, chemotherapy | Not supported with pagination yet | null |
PMC7305372_01 | Male | 28 | A 28-year-old male was seen by the digestive surgery for a lump on the right groin area with a back pain (VAS 5) for the past four months. Because there was no abnormality in abdomen area, the patient was consulted to Orthopaedic Department. The pain did not radiate and was relieved when the patient lies down on his back. There was no numbness or tingling sensation. He had no trouble with micturition and defecation. The patient did not have a history of lung tuberculosis. However, the patient was sent to the pulmonary division before our department and was diagnosed with tuberculous spondylitis and was given anti-tuberculous drugs since then. The clinical outcomes were assessed preoperatively and at the final follow-up by Oswestry Disability Index (ODI) and Japanese Orthopaedic Association (JOA) scores. The preoperative ODI score for this patient was categorized as moderate disability, whereas the JOA score for this patient is in normal function state. | abscess evacuation, arthroscopy, debridement, lumbosacral tuberculosis, minimally invasive surgery | Not supported with pagination yet | null |
PMC4899672_03 | Male | 45 | Patient 3:In June 2010, a 45-year-old brother of patient 2 presented with transient right-arm and hand weakness, and numbness with dysarthria for one day. He subsequently developed staring episodes thought to represent seizure activity, headaches, and behavioral changes including inappropriate behavior in public places, disinhibition, and anger outbursts. Brain MRI on a 3.0 T magnet (Fig. 5) revealed multiple cortical and subcortical areas of blood products bilaterally involving the supratentorial cerebral hemispheres. One large lesion in particular affecting the cingulum was thought to contribute to the behavioral issues. | ct, computed tomography, mri, magnetic resonance imaging | Not supported with pagination yet | null |
PMC6327707_01 | Female | 48 | A 48-year-old female presented to us with a history of symmetric, tingling paresthesia in the lower limbs for the past 9 months, which progressively ascended to involve the trunk and neck. On examination, she had loss of joint position and vibration sensations over the lower limbs and trunk, a sensory level at the mid-thoracic region, spasticity of bilateral lower limbs, and a positive Romberg's sign. MRI spine and brain showed longitudinally extensive T2 and fluid-attenuated inversion recovery hyperintensities, involving the posterior column with contrast enhancement [Figure 1]. MRI brain was normal. Causes of longitudinally extensive tract specific spinal cord lesions including neuromyelitis optica spectrum disorders (NMOSDs), paraneoplastic myelopathies, and Vitamin B12 deficiency were initially considered. Laboratory tests revealed normal levels of serum Vitamin B12 levels (1085 pg/ml; normal range 211-911 pg/ml) and serum copper (115 mug/dl; normal range 85-155 mug/dl) whereas serum aquaporin 4 antibody was negative. Screening for blood biomarkers for cancer including cancer antigen (CA)-125, Carcino Embryonic Antigen, CA 19-9, and alpha-feto protein were negative. Cerebrospinal fluid examination revealed five cells (all lymphocytes), mildly elevated proteins (57 mg/dl; normal 15-45 mg/dl), and normal sugar (66 mg/dl; normal 45-80 mg/dl). Computerized tomography (CT) of the thorax showed paratracheal, hilar, and subcarinal nodes in addition to the right upper lobe fibrosis. Serum angiotensin-converting enzyme levels were normal (50 U/L; normal range 8-53 U/L). CT-guided transthoracic biopsy of the mediastinal lymph nodes was then performed and histopathologic evaluation revealed noncaseating granulomas. Staining and polymerase chain reaction for mycobacterium tuberculosis was negative. A diagnosis of systemic sarcoidosis with neurological involvement was made, and the patient was started on intravenous followed by oral steroids. She noted significant improvement in clinical symptoms with treatment. | null | Not supported with pagination yet | null |
PMC7512063_01 | Male | 60 | The 60-year-old male patient came to the chest diseases department of our hospital with the complaints of progressively increasing cough, dyspnea, and fatigue in the recent two years. The patient who denied hemoptysis said that he rarely produced sputum. He had no chronic disease except AS diagnosed in 1976. He never smoked. He had no alcohol consumption. He had no continuous medication except nonsteroid anti-inflammatory drugs (NSAIDs) for 2-3 days monthly since his pain of the musculoskeletal system decreased dramatically in the recent years. His physical examination revealed a blood pressure of 110/70 mmHg and 36.8 C body temperature. Heart beats were rhythmic, and no murmur was present. Breath sounds decreased in the upper zone of the right lung. The chest expansion and the lumbar Schober test measured 0 cm. Laboratory test results were found as follows: erythrocyte sedimentation rate (ESR) 72 mm/h, C-reactive protein (CRP) 28 mg/dL (normal range: 0-5), leukocytes 7800/mm3, hemoglobin 11 g/dL, platelets 323.000/mm3, and total protein/albumin ratio 6.8/3.3 g/dL. Other biochemical tests, electrolytes, and urinalysis results were within normal limits. Spirometric pulmonary function tests (PFT) revealed a restrictive pattern: a forced vital capacity (FVC) of 1.58 L (38% of predicted), a forced expiratory volume in 1 s (FEV1) of 1.50 L (46% of predicted), and an FEV1/FVC 94% and carbon monoxide diffusion capacity (DLCO) of 23 mL/min per mmHg (43% of predicted). Lung radiography revealed increased diffuse opacity in the upper zones of the right lung (Figure 1). Also, it showed right tracheal deviation and blunting of the right costophrenic sinus. Thoracic HRCT revealed the chronic consolidation with traction bronchiectasis compatible with fibrosis in the apical and posterior segments of the right upper lobe extending to the superior segment of the right lower lobe (Figure 2). In addition, there were also nodular alveolar densities in the lateral basal segment of the right lower lobe. Thoracic CT performed in 2016 showed mild bronchiectasis and sequela parenchymal bands in the middle lobe of the right lung. However, there was no apical fibrotic change, traction bronchiectasis, and parenchymal destruction in the upper lobe of the right lung (Figure 3). The comparison between thoracic HRCT and CT images suggested that pulmonary apical fibrosis and parenchymal destruction encountered in the right lung have developed in the recent three years. Gram staining, methylene blue stain, and Ehrlich-Ziehl-Neelsen (EZN) stain of the sputum revealed no pathogen, and all subsequent sputum cultures were negative. The tuberculin skin test resulted 0 mm, and the interferon-gamma release assay (IGRA) detected no Mycobacterium tuberculosis infection. Bronchoscopy and transbronchial lung biopsy were performed. Biopsy did not reveal any finding of malignancy, granulomatous inflammation, or vasculitis. Lumbar vertebral and pelvis X-ray revealed bilateral grade IV sacroiliitis, syndesmophytes, and appearance of "bamboo spine." As a conclusion, the patient was diagnosed with pulmonary involvement of AS including apical pleural thickening with upper lobe fibrosis and nonspecific interstitial abnormalities. Etanercept 50 mg/week and meloxicam 15 mg/day were administered. The patient was hospitalized for two weeks and included in the pulmonary rehabilitation programme. The next control examination after six weeks resulted as follows: ESR 36 mm/h, CRP 8 mg/dL, FVC of 41% of predicted, FEV1 of 49% of predicted, FEV1/FVC 96%, and DLCO 71% of predicted. After a six-month course of etanercept treatment, the patient did not develop new pulmonary lesion, and the drug was well tolerated without any adverse events. | null | Not supported with pagination yet | null |
PMC10032216_01 | Male | 44 | A 44-year-old male patient complained of "a 20-day history of neck pain with quadriplegia for 2 days". Cervical spine CT (Figure 1A-D) at a local hospital showed abnormalities in his C6-7 cervical segment, and conservative treatment was performed with poor results. Two days before admission, he experienced numbness and weakness in his limbs, difficulty urinating, and a fever with a body temperature up to 38 C. Cervical magnetic resonance examination found: C6/7 abnormal signals of the vertebral body and intervertebral disc, abscess formation under the anterior longitudinal ligament and spinal canal, compression of the spinal cord, and infectious lesions (Figure 2A-C). On the day of admission, the physical examination showed limited flexion and extension of the cervical spine, weak bilateral upper extremity muscle strength (bilateral triceps muscle strength class I, bilateral biceps muscle strength class II, grip strength grade 0), and loss of tendon reflexes. Both lower limb muscles had a strength level of 0 and low muscle tone. Additional physical examination findings included: bilateral Achilles tendon reflex (-), knee tendon reflex (-), Hoffmann's sign (-), sphincter contraction without touch, bilateral ankle burn, heavy right side, and second-degree scald (Figure 3A and B). Laboratory tests, including the Brucella antibody test, tuberculosis antibody test, and Mycobacterium tuberculosis T-cell test, were negative, and serology showed inflammation (Table 1).
Two days after admission, the patient underwent posterior cervical spine decompression and fixation under general anesthesia. Preoperative X-ray examination showed C6-7 cervical vertebral body abnormalities and cervical spine degeneration (Figure 4A and B), and the operation went smoothly and the decompression was adequate. Postoperative examination of the internal fixation position was good, as seen in perspective (Figure 4C and D), but there was no significant improvement in limb mobility immediately after the operation. On the first day after surgery, the patient indicated a significant recovery in the strength of both upper limbs, and the physical examination showed that the grip strength of both hands was restored to grade 3 and the sensation of both lower limbs was restored, yet the muscle strength was not significantly improved. On the second day after surgery, the patient complained of an increase in body temperature at night, up to 38.5 C, and an increase in his inflammatory factors compared to preoperatively. Blood culture findings showed Streptococcus constellatus infection, and antimicrobial susceptibility test results indicated that blood isolates were sensitive to vancomycin, penicillin G, cefepime, cefotaxime, levofloxacin, chloramphenicol, and linezolid. Considering that the patient had severe symptoms, the patient was given systemic antibiotic therapy (vancomycin injection 1000 mg, q12h, IV drip; Cefzoxime sodium 1 g, q8h, IV drip) and symptomatic treatments such as fluid replacement, maintenance of water-electrolyte balance, and nutritional support.
After 2 weeks of vancomycin and cephalosporin treatment, the patient's blood culture reexamination results were negative, the serological inflammatory indicators showed a downward trend (Table 1), the body temperature remained stable at 38 C, and the sensory and muscle strength of both lower limbs recovered more than before, the left side was obvious and the right side was weak (Supplementary Video 1); however, there was a neck rash. The dermatology department and clinical pharmacy department considered that the neck rash was related to the previous use of vancomycin, so we changed the antibiotic (linezolid 0.6 g, q12h, IV drip) for symptomatic treatment. After 3 weeks of systemic antibiotic treatment, the patient's condition tended to be stable, and the muscle strength and sensory motor function of both lower limbs were significantly improved compared with those before surgery (Supplementary Video 2). The muscle strength of the left lower limb was grade 4, and that of the right lower limb was grade 3. Inflammatory indicators showed a good downward trend (Table 1). The MRI reexamination results showed that the cervical spine infection was relieved compared with the previous observation (Figure 5A-D). CT reexamination of the cervical spine showed that the C6/7 vertebral body was severely damaged and the bone defect was obvious (Figure 6A-F). Therefore, the patient underwent a second operation under general anesthesia for an anterior infection lesion removal and internal fixation of the cervical spine. During the operation, it was found that the lower edge of the C6 vertebral body, the upper edge of the C7 vertebral body, and the C6/7 intervertebral space were obviously damaged, cavities were formed, and some blood gushed out of the cavity (Figure 7A-C). The infection lesion was carefully removed, and the tissue was removed for routine pathological culture. A fibular allogeneic bone of appropriate length (Figure 7D and E) was selected and placed between the C5-7 vertebral bodies, and an anterior cervical titanium plate was fixed in front of C5 and C7 (Figure 7F). The results of pathological culture during the operation showed acute and chronic inflammatory cell infiltration and granulation tissue formation, and the clinical results were consistent with inflammatory lesions (Figure 8A and B). According to all the test results, tuberculosis infection and Brucella infection were excluded, and the patient was finally diagnosed with SEA caused by Streptococcus constellatus.
Within a week of the second operation, the patient continued to receive systemic antibiotics, and the serological inflammatory index gradually decreased to normal. Bacterial culture and anaerobic culture of the puncture fluid were both negative (Table 1), indicating that systemic antibiotic therapy was effective. The patient's CT (Figure 9A-D) and X-ray (Figure 9G and H) review images showed that the internal fixation position was good, the intervertebral space and C6-7 vertebral bone mass were filled on the CT review image (Figure 9E and F), and the patient was discharged. Two weeks after discharge, the patient's serological examination showed that the inflammatory indicators tended to be normal, and there was no significant change (Table 1). | streptococcus constellatus, diagnosis and treatment, epidural abscess, pyogenic spondylitis, vertebral destruction | Not supported with pagination yet | null |
PMC4900063_01 | Female | 41 | This 41-year-old female had a sudden onset of bilateral ankle pain in 1993, after playing basketball. She was unable to walk without pain for a week, and sought medical care. She said that radiographs at that time showed "small fractures" and "dead cartilage" in her ankles. Her pain improved gradually over the next six months with conservative care, but then began recurring more and more frequently, interfering with her previously active lifestyle of running and playing tennis. She had no history of ankle trauma.
Over the same time period, she also developed pain, stiffness and swelling of her fingers. This was considered to represent seronegative rheumatoid arthritis. These symptoms have been well-controlled with a low carbohydrate, no sugar diet. She had not taken steroids for this.
The patient was born in Northeast China, and reported that a number of family members and friends from Northeast China have had similar symptoms. She was concerned that she and they have Kashin-Beck disease.
Her ankle pain became progressively worse over the next 12 years, and in 2005 she sought podiatric care. Radiographs revealed osteochondral defects of both talar domes (Fig. 1).
Magnetic resonance imaging (MRI) of both ankles showed osteochondral irregularity and collapse in both talar domes (Fig. 2).
Computed tomography (CT) of both ankles was then performed to assist with surgical planning, and also showed osteochondral irregularity and collapse in both talar domes (Fig. 3).
She was then seen by an orthopedic foot and ankle specialist, who discussed several treatment options, including talar-dome drilling and bone grafting, osteoarticular allograft, total ankle arthroplasty, and ankle arthrodesis. At present, our patient is continuing conservative care, but is considering eventual osteoarticular allografts. | ct, computed tomography, mri, magnetic resonance imaging | Not supported with pagination yet | null |
PMC9874059_01 | Male | 36 | A 36-year-old lifetime non-smoker obese man with no history of smoking was referred to our office due to a 9-year history of progressive exertional dyspnea and yellow productive cough accompanied by occasional hemoptysis. He reported being able to climb two flights of stairs before becoming dyspneic. His medical history was significant for childhood asthma, hypertension, and impaired glucose tolerance. Outpatient medications included Lisinopril 10 mg daily orally. He works as a security guard in a hospital five days per week and is relatively high functioning. He denied any workplace exposure or alcohol consumption. He migrated from Cuba at the age of seven. And there was no relevant family history.
Vital signs and physical exam were unremarkable. Initial chest radiograph (Fig. 1) showed innumerable pulmonary nodules in both lungs, up to 4 mm in greatest dimension, predominantly involving the middle to lower lung fields. Findings were concerning for miliary tuberculosis (TB) given a history of positive purified protein derivative (PPD) skin test, which might also be attributed to Bacille Calmette-Guerin (BCG) vaccination during childhood. High-Resolution Computed Tomography (HRCT) (Fig. 2, Fig. 3) showed innumerable pulmonary nodules scattered throughout the lungs and along the broncho-vascular bundles measuring 2-5 mm. Differential diagnosis included miliary TB, granulomatous diseases, pneumoconiosis, certain deposition diseases (such as amyloidosis), or alveolar microlithiasis in the appropriate clinical setting. The findings also were suggestive of dystrophic calcifications in the setting of scarring from prior infection/inflammation. A presumptive diagnosis of pulmonary alveolar microlithiasis (PAM) was at this time presumed, given radiographic progression and clinically indolent presentation. Pulmonary function tests (PFT) revealed a total lung capacity (TLC) of 5.37 L (89% predicted), forced vital capacity (FVC) 3.57 L (87% predicted [normal >=80%]), forced expiratory volume in 1 second (FEV1) 3.19 L (94% predicted [normal >=80%]), FEV1/FVC 90% (normal >=70%), diffusing capacity for carbon monoxide (DLCO) 69% (normal >=70%). In summary, normal lung volumes and a mild isolated decrease in DLCO was found.
Bronchoscopy, bronchoalveolar lavage, and transbronchial lung biopsy revealed no abnormalities. The Quantiferon TB test was negative, ruling our active or miliary TB. Histoplasma antibody test was also negative. Genetic testing for SLC34A2 mutation was negative, lowering suspicion for PAM. The patient subsequently underwent a right video assisted thoracoscopic surgery (VATS) (Fig. 4) and segmentectomy of the basilar segment of the right lower lobe, the medial segment of the right middle lobe, and the anterior segment of the right upper lobe without any complications.
Upon gross examination, palpation of the specimen revealed multiple firm sub-pleural nodules. Sectioning through the specimen revealed innumerable calcified nodules. Microscopically (Fig. 5, Fig. 6), diffuse ramifying spicules of mature woven bone arranged in a serpentine fashion along the alveolar septa associated with fatty bone marrow were identified, confirming the diagnosis of DPO. Although bone marrow was present, extensive sampling failed to reveal hematopoiesis. Of note, there was no evidence of significant underlying pathologic findings, such as interstitial fibrosis, granulomas, organizing pneumonia, or significant inflammation in the background lung parenchyma. | case report, diffuse pulmonary ossification, nodular pulmonary ossification, pulmonary alveolar microlithiasis | Not supported with pagination yet | null |
PMC4329767_01 | Male | 26 | We report the case of a 26-year-old male who presented to us with the complaint of fever for 7 days which was insidious in onset, low grade without chills, and rigors. He also noted diarrhea for 5 days which was watery, 4-5 times per day associated with diffuse abdominal discomfort but not associated with the passage of blood or mucous. He then noticed jaundice and passage of dark urine for 3 days without any decrease in the frequency or volume of urine. There was no prior history of blood in urine or cola colored urine or burning micturition. There was no history of jaundice, pruritus, clay stools, melaena, hematemesis, abdominal distension, or altered sensorium. He reported only an occasional intake of ethanol. The patient denied intake of indigenous medications or intoxication. The patient did not report any past major surgeries, blood transfusions, or IV drug abuse prior to onset of the disease. He did not report any history of diabetes, hypertension, tuberculosis, thyroid disease, trauma, exposure to industrial toxins or radiation, blood or blood component therapy, bleeding disorders, promiscuity, or similar complaints in the family or neighbourhood. At the time of admission, he was conscious, was oriented, and was febrile. His blood pressure was 110/70 mm of Hg in the right arm, pulse-108/min. He was icteric and did not have pallor, clubbing, cyanosis, pedal edema, or lymphadenopathy. He did not have any skin rash or stigmata of chronic liver disease such as spider angioma, palmar erythema, and parotid enlargement. His initial lab data revealed a hemoglobin of 12 g/dL, total leucocyte counts 11330/dL, and INR (international normalized ratio) of 2.69. His serum bilirubin was 4.5 mg/dL with predominant direct fraction of 3 mg/dL and indirect fraction of 1.5 mg/dL. Liver enzymes showed aspartate transaminase (AST) 452 IU/L, alanine transaminase (ALT) 2750 IU/L, alkaline phosphatase 254, and gamma glutamyl transferase (GGT) 169. Serum albumin was reduced at 2.2 g/dL with globulins 2.7 g/dL. His blood urea was 132 mg/dL with serum creatinine levels of 9.37 mg/dL with normal serum electrolytes suggestive of acute kidney injury. His IgM anti-HEV was positive and serology for hepatitis A, hepatitis B, hepatitis C, HIV, dengue, and Leptospira was negative. His peripheral blood smear and rapid malaria test was negative. Due to his deranged renal functions, he was started on slow low efficiency dialysis (SLED) sessions and gradually his urine output improved over the next few days. He was managed conservatively with IV antibiotics, IV fluids, nutritional therapy, and other supportive measures. After two sessions of SLED, he progressed to a polyuric phase, so dialysis was stopped and patient was managed conservatively. On day 6 of presentation, he developed fever again with left sided pleuritic pain and sudden onset of shortness of breath. He was found to be restless and dyspneic and on auscultation he had an S 3 gallop rhythm. Due to increasing respiratory distress, he was intubated and required mechanical ventilation for 3 days. Arterial blood gas analysis revealed type 1 respiratory failure with hypoxemia, and ECG showed sinus tachycardia and dynamic ST-T changes. Chest X-ray was suggestive of pulmonary congestion. Urgent 2D echocardiography revealed global hypokinesia, with normal LV and RV size, but ejection fraction was reduced to 25-30% with preserved right ventricular function. Troponin I was positive at 0.5 ng/mL and creatinine kinase-MB fraction levels were increased at 68 IU/L, 28% of total CK. This was suggestive of myocarditis. He was followed up by daily echocardiograms (see Figure 1). Over a period of 2 days, he gradually improved and was weaned off mechanical ventilation. His ejection fraction improved to 45% by day 3 and 60% with normal ventricular function by day 6. We performed a cardiac magnetic resonance imaging study on day 10 of myocarditis, but by then there was only marginal hypokinesia of the lateral left ventricular wall (see Figures 2 and 3). He was discharged on day 11 of hospital stay. | null | Not supported with pagination yet | null |
PMC3869631_01 | Male | 83 | An 83-year-old Japanese man had suffered from productive cough for 6 months and visited our hospital in March 2011. He had no fever. His sputum was yellowish. He had smoked 60 pack-years until 50 years of age. He had no past history of illness. He was a tobacco farmer and had also raised silk-worms. On physical examination, there was no lymphadenopathy, no clubbing, and no edema. SpO2 was 98% and blood pressure was 150/80 mmHg. His vesicular sound was slightly decreased throughout both lung fields and no crackle was auscultated. A chest X-ray and computed tomography (CT) scan showed emphysematous changes and multiple cavities in the bilateral pulmonary fields (Figure 1). Laboratory test results were as follows: WBC, 6500/mul; RBC, 3.29 million/mul; hemoglobin, 9.9 g/dl; MCV, 94.5 fl; MCH, 30.1 pg; total protein, 6.8 g/dl; albumin, 3.0 g/dl; total cholesterol, 167 mg/ml; BUN, 29.4 mg/ml; Cr, 1.57 mg/ml; and CRP, 1.34 mg/ml. Urine examination showed a sub-nephrotic range of proteinuria (3.1 g/gCr) and many hyaline casts (>50/total fields) in the urinary sediment. Pulmonary function tests showed low%VC (70.8%) and normal range of FEV1.0% (78.1%). Acid-fast bacilli were evident in his sputum by Ziehl-Neelsen staining (Gaffky 3). PCR amplifications for Mycobacterium tuberculosis, Mycobacterium avium, and Mycobacterium intracellulare were all negative. Numerous colonies of acid-fast bacilli appeared upon culture. DNA-DNA hybridization, which can detect 18 species of acid-fast bacilli using cultured colonies, failed to identify the species. The mycobacterial characteristics of a sputum sample obtained on another day were exactly the same as those of the first sample. Finally, Mycobacterium shimoidei was identified by rpoB sequencing and 16S rRNA sequencing, with 100% homology to the sequences.
Renal tissue samples obtained by renal biopsy mainly showed membranous glomerulonephritis (MGN), with partial hypertensive renal damage (Figure 2). In addition, the mesangial areas were slightly enlarged and mesangial cells had proliferated in part (Figure 2A). Electron-dense deposits were observed in the mesangial areas as well as the subepithelium and basement membranes (Figure 2C). Immunofluorescence analyses showed positive staining for IgG, IgA, and IgM in the basement membranes (data not shown). No granulomatous change was observed in the renal tissue samples. No acid-fast bacilli were detected by Ziehl-Neelsen staining.
Because the productive cough continued, M. tuberculosis was excluded, and a new lesion appeared on his chest X-ray, anti-mycobacterial treatment (clarithromycin, 400 mg/d; rifampicin, 300 mg/d; and ethambutol, 500 mg/d) was started before the identification of M. shimoidei. Candesartan (80 mg/d), an angiotensin II receptor blocker (ARB), was also started. After 1 month of treatment, the species was identified as M. shimoidei and the initial treatment was continued. The isolate was sensitive to ethambutol, streptomycin, kanamycin, enviomycin, ethionamide, and levofloxacin, and was resistant to isoniazid, rifampicin, cycloserine, and para-aminosalicylic acid, in in vitro drug sensitivity tests. At 2 months after starting the treatment, his sputum was clearly decreased. After 6 months of treatment, acid-fast bacilli were not detected by either Ziehl-Neelsen staining or 8-week culture, and the chest CT findings were also improved (data not shown). He continued antimycobacterial treatment for an additional 1 year (18 months in total) in accordance with the statement from the American Thoracic Society and the Infectious Diseases Society of America. The amount of urine protein decreased gradually after initiation of the antimycobacterial treatment and ARB, and reached 0.6 g/gCr. The serum albumin level recovered to 3.6 g/dl at the end of the treatment. | membranous glomerulonephritis, mycobacterium, non-tuberculous, proteinuria, shimoidei | Chest X-ray and computed tomography. (A) Chest X-ray showing cavities (arrow), nodules, and pleural thickening (arrowhead) in the bilateral, predominantly upper, lung fields. |
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PMC4756770_01 | Male | 7 | A 7-year-old white male with a 3-year history of anxiety and obsessive-compulsive behavior presented to a multi-physician suburban integrative medicine practice. The patient's mother reported that when the boy was 2 years old, she started receiving daily incident reports from his daycare of his pushing other children and violent outbursts. A child psychologist evaluated his behavior as normal. At age 3.5 years, the child began to be fearful and dependent. He started expressing severe separation anxiety, refusing to enter any room alone, including the bathroom. He was increasingly irritable and had trouble interacting with other children. When the patient was 5 years of age, his teacher noted that he had obsessive thoughts about "germs" and developed compulsive hand-washing. He avoided going near trash cans because he was afraid of "germs." He expressed fear of his feces and would only go to the bathroom if he were completely undressed. He expressed irrational fears of getting "lead poisoning" and developed a related phobia toward pencils, to the extent that he was unable to sit at a table where a pencil was present. He did not want to play with other children because he was scared that he would hurt them. Whenever he coughed from a mild upper respiratory infection, he feared that he was going to stop breathing and became preoccupied with death. The patient was taken to a child psychologist and diagnosed with OCD and anxiety disorder. He was started on cognitive behavioral therapy, which included play therapy, exposure therapy, and other goal-directed therapy that resulted in mild improvement of his condition.
Positive for nervousness, sadness, depression, irritability, anxiety, and difficulty concentrating. The patient had also been diagnosed with spatial apraxia, which resulted in the patient bumping into other children that precipitated conflicts. Finally, the patient had a pruritic rash which started at age 1 shortly after he started to eat solid foods. The rash was worse in the summer and located behind both elbows and both knees. It was persistent despite various lotions, topical steroid creams, and special baths.
Prolonged labor (>36 hours); chronic ear infections starting at age 3 months; Clostridium difficile colitis related to amoxicillin treatment; serum sickness after a course of antibiotics.
None. Patient's mother did not wish to do a trial of medication in light of the patient's episodes of rash, Clostridium difficile colitis, and serum sickness.
Patient was receiving occupational therapy for his spatial apraxia.
Multivitamin, docosahexanoic acid, fish oil supplements
Penicillin (rash), cephalosporin (rash).
Melanoma (mother), anxiety disorder (paternal aunt).
Anxious-appearing boy who appeared his stated age sitting next to his mother. Patient had a restricted affect and displayed little eye contact with the examiner. He displayed no psychotic symptoms. There were no unusual thought processes observed. There was an erythematous rash on the popliteal fossa, elbows, and chest.
Autonomic response testing (ART) was performed to determine the targets of therapy and guide which areas of the body and ear acupuncture points should be treated with low-level laser therapy (LLLT). ART was positive for wheat, barley, and rye (gluten sensitivity), lead, mercury, and Entamoeba.
Table 1 provides an overview and time-line of the patient's treatment course. Over the course of 5 visits in a 5-month period, the patient was treated with the following: (1) a gluten-free diet and avoidance of gluten-containing personal care products such as soaps, shampoos, toothpaste, and hair products; (2) low-level laser ear acupuncture using a 904 nm laser RJ Olympic Physiolaser (RJ Laser, New Brunswick, Canada) set at 150mW for 15-second pulses using various Nogier frequencies; (3) LLLT using a 532 nm 5 milliwatt green pen laser pointer to reduce sensitivity to or amount of heavy metals:per ART indication, treatment was applied to hairy portions of the head for 3 minutes with either EDTA, dimercaptopropanesulfonic acid (DMPS), or wheat adjacent to the laser beam; (4) ionic foot baths during the office visit to reduce the total body burden of heavy metals; (5) low-dose chelation therapy using DMPS 100 mg twice per week; and (6) microcurrent therapy to address a subclinical Entamoeba infection.
The patient's mother reported that the patient's OCD symptoms and anxiety improved 40% since the previous visit. She noticed that her child was less anxious and more at ease in social situations and did not bring up his fear of his stool. She also reported that his eczema improved 90%, which was a greater improvement than with any other treatment previously tried. She admitted that while the majority of her son's diet was gluten free, there were a few occasions where her son had gluten intake. She also reported that the patient had not started to use gluten-free personal care products. The mother was unable to determine any differences in OCD and anxiety symptoms the day of or after gluten intake.
The patient's mother reported that her son's OCD symptoms had improved 80% and his anxiety had improved 90% since the last visit. She stated that he generally had more energy during the day. His eczema was 100% improved. The patient ate only gluten-free food and used gluten-free personal care products.
The patient's mother reported that her son's OCD improved 75% and his anxiety had improved 85% overall. He had more energy. However, he still had trouble reading and focusing on words.
The patient's mother reported that her son's OCD and anxiety symptoms were 99% better. He no longer had trouble staying in a room by himself. Repetitive behavior, such as hand washing and hand sanitation, had resolved. He no longer showed any fear of his feces, and was able to go to the bathroom without taking off all his clothes. The patient was 100% free of eczema.
The patient's mother reported her son was doing significantly better since the start of the integrative medicine therapies. She reported 1 instance of a relapse in symptoms of irritation and anxiety the day after the child had eaten food that had been cross-contaminated with food containing gluten from another family member. In general, she reported that he was less anxious, did not worry about his health or dwell on death, did not express concern about the trash can or germs, could go to the bathroom without completely undressing, had friends, and was less socially fearful. However, she did report that in the past month, he had begun to echo his words by mouthing them to himself and continually inquires about the time and the exact length of activities. While the patient had drastic improvements with the treatments, the patient still needed ongoing treatment.
Patient's mother was very satisfied with patient's improvement symptom-wise, socially, and academically. The palilalia resolved. Whereas the patient had failed his kindergarten entrance test, now in second grade he scored higher than his grade level on standardized testing. The patient continued to follow a strict gluten-free diet and to avoid gluten-containing personal care products.
Patient's mother reported that her son had not had any recurrent or new symptoms. She reported compliance with the gluten-free regime. When inadvertent gluten exposure from family members occurred, the patient did demonstrate some symptomatology, which rapidly resolved. The mother reported that she occasionally used the laser pointer pen for home LLLT. She did this at times of inadvertent gluten exposure. Her son was no longer in individual or group therapy and was on no medications. His psychologist confirmed his improvement. Mother reported also that the patient's spatial apraxia resolved and he no longer needed occupational therapy. She stated the apraxia markedly improved concurrently with the OCD improvement.
The patient's mother paid for all treatment costs as none of the treatments was covered under her insurance. Total treatment costs were less than $1000 (Table 2). | anxiety, dmps, gluten, heavy netal body burden, lead chelation, mercury, obsessive-compulsive disorder | Not supported with pagination yet | null |
PMC8093052_03 | Female | 26 | A 26-year-old woman presented with a six-year history of dry eyes and dry mouth, shortness of breath on exertion, and fatigue. She was in active military service at the time. Prior to the onset of shortness of breath, she had been in excellent physical health. Six years earlier, she had woken up with severe conjunctival injection attributed to a corneal abrasion. She was evaluated by an ophthalmologist and Schirmer's test was positive bilaterally. She received a presumptive diagnosis of primary Sjogren's syndrome confirmed by minor salivary gland and lip biopsy. She was treated with pilocarpine eyedrops for keratoconjunctivitis sicca. A year prior to visiting our center, she developed recurrent episodes of bronchitis complicated with pneumococcal pneumonia and sepsis. Since then, she had persistent shortness of breath. Two-dimensional echocardiogram did not show pulmonary hypertension. She had mild heartburn and an esophagram showed moderate esophageal dysmotility. No evidence of aspiration was found on a tailored barium swallow. She denied having Raynaud's phenomenon.
Pulmonary function tests demonstrated largely normal lung volumes with a TLC of 4.58 (95% predicted), reduced FEV1 of 2.39 L (77% predicted) and FVC of 2.89 L (80% predicted), and preserved FEV1/FVC ratio of 95% without significant bronchodilator response. DLco was reduced at 17.74 mL/min/mmHg (62% predicted). Serum protein electrophoresis and immunofixation did not demonstrate IgG kappa monoclonal protein.
Laboratory abnormalities included a very high titer ANA 1 : 5120 speckled pattern, elevated ENA profile at 121 units (normal value: <20 units), SSA/Ro 60 at 114 units, SSA/Ro 52 at 120 units (normal value: <20 units), SSB/La at 148 units (normal value: <20 units), and RF at 20 IUs/mL. Immunoglobulin A (IgA) and IgG were elevated at 1104 mg/dL (normal range: 50-462 mg/dL) and 3956 mg/dL (normal range: 700-1620 mg/dL), respectively, while IgM and IgG were within the normal limits. C4 was decreased at 18.3 mg/dL (normal range: 19-52 mg/dL). She had mild normocytic normochromic anemia, hemoglobin of 12.1 g/dL and hematocrit of 36%, with elevated inflammatory markers erythrocyte sedimentation rate at 69 mm/hr and CRP within normal limits.
An HRCT scan of the chest prior to our evaluation demonstrated pulmonary nodules, thin-walled cysts, basilar predominant ground glass attenuation, and subpleural consolidation (Figure 4(a)). Repeat HRCT demonstrated increased ground glass attenuation, bilateral solid pulmonary nodules, a few of which had slightly increased in size, and stable thin-walled cysts. The pattern was consistent with lymphocytic interstitial pneumonia (LIP) in the setting of Sjogren's syndrome (Figure 4(b)). Amyloidosis and lymphoproliferative disorder/lymphoma remained additional considerations for the solid nodules, which had increased in size.
The patient underwent CT-guided lung biopsy of the right lung upper lobe. This was unremarkable for malignancy; however, immunohistochemistry was consistent with light chain amyloid, while the kappa light chain staining was the strongest (Figure 5). There was significant lambda staining, but a clear monoclonal gammopathy was not identified. Amyloid staining did not confirm amyloidosis, and a Congo red stain was not detected.
The patient was initially treated with corticosteroid taper. She complained of symptoms consistent with neuropathy as well as constant vertiginous symptoms and visual disturbances of diplopia and intermittent pressure headache. Electromyography (EMG) and nerve conductive velocity (NCV) studies were within normal limits. She was found to have acetylcholine receptor antibodies.
The patient remained an active-duty soldier. We treated her for LIP with rituximab 1000 mg, 2 doses, 14 days apart every 6 months. Later, IVIG was added to her regimen to address nervous system involvement. Her exercise tolerance improved and DLco normalized (101% predicted).
In LIP, lymphocyte and plasma cells are diffusely infiltrated in the lung parenchyma, along with polyclonal plasma cells and lymphocytes. Elevated immunoglobulins IgA and IgG can be the results of polyclonal B cells. On three occasions, the patient's serum protein electrophoresis showed polyclonal gammopathy and on one occasion it showed monoclonal gammopathy. These gammopathies resolved with rituximab therapy, as did the level of immunoglobulins and RF and sedimentation rate. Follow-up HRCT showed an overall improvement in lung involvement characterized by a decrease in ground glass opacities and the solid component of nodules with stability of cysts. We could not exclude that the patient had a low-grade pulmonary lymphoma that responded to rituximab. | null | Not supported with pagination yet | null |
PMC7677661_01 | Unknown | 59 | 59-year-old patient, rural resident, who lives in a poor region of Morocco endemic to tuberculosis., menopausal for 5 years, without particular pathological history, was not taking any drugs and with no medical family history, consults for swelling in the right breast The onset of symptoms goes back 5 months by autopalpation of a nodule in the right breast associated with a progressive increase in its volume associated two months later with a mastodynia, without nipple discharge, without local inflammatory sign.
Clinical examination shows 2 masses occuping the entire upper outer quadrant of the right breast, respectively 8 cm, 4 cm in diameter and prolapsing in the axillary region, of soft consistency, well limited, mobile and painless, without inflammatory phenomenon of the skin integuments opposite. There is no axillary or supraclavicular lymphadenopathy, nor nipple discharge (Fig. 1).
Radiological explorations (mammography + ultrasound) (Figs. 2, 3) carried out objectified:
the first measures 66 x 84 mm in transverse diameter and has a thin wall and anechoic content. classified ACR2 BIRADS.
The second measures 49 x 47 mm in transverse diameter in the axillary hollow and has ultimately echogenic content with a sloping portion producing the appearance of pseudo vegetation. classified ACR3 BIRADS.
A voluminous opacity of the QSE of the dense and well limited right breast corresponding on ultrasound to two cystic masses of the QSE and the right axillary hollow.
The two masses are not vascularized by Doppler.
An abdominal ultrasound, a chest x-ray, as well as a biological blood test (complete blood count, ionogram) performed are completely normal. A fine needle aspiration of the cyst only showed the presence of altered polynuclear, without evidence of tumor cells. Treatment consisted of surgical excision such as perikystectomy (Fig. 4) who was performed by junior resident with 5 years of specialised training, and pathology examination demonstrated breast hydatidosis.
Postoperative evolution was favourable. Patient was put on medical treatment based on albendazole immediately. The patient was monitored for one year without local or distant recurrences. | breast, breast cyst, hydatid cyst | Not supported with pagination yet | null |
PMC9924618_01 | Female | 23 | We present a 23 year old female from Micronesia who presented as a transfer from Bowling Green, KY facility due to sepsis and abdominal pain. The patient had a previous admission for abdominal pain one year ago. A CT abdomen performed at that time was significant for what appeared to be widespread carcinomatosis. A biopsy was performed and pathology reports were significant for non-caseating granulomas, rather than occult malignancy. AFB and fungal stains were negative. Patient failed to follow up after this evaluation.
The patient then presented to Bowling Greene with a significant fever and recurring abdominal pain. A CT abdomen/pelvis was notable for a R. lower lobe infiltrate, left bibasilar atelectasis, a small pleural effusion, and lymphadenopathy in the mediastinum, abdomen, retroperitoneum, and pelvis. The patient was transferred to our facility for higher level of care. On presentation, labs were significant for a lactic acidosis and leukocytosis, with the patient requiring vasopressors. The patient was started on broad-spectrum antibiotics for the treatment of acute sepsis. ID and general surgery were consulted. General surgery recommended medical management only. Fungal, viral, and sarcoidosis work ups were started, per ID. The patient was placed on prophylactic steroids and a repeat Quantiferon was ordered. Both fungal and viral testing returned negative while the patient's quantiferon gold test returned positive. A CT chest with contrast was ordered for further work up of adenopathy with concern for TB after the positive quantiferon gold. Imaging was significant for multiple, small mediastinal lymph nodes and diffuse tree-in-bud findings throughout the lung parenchyma. Pulmonology was consulted for a bronchoscopy with EUS-guided biopsy. Pathology revealed necrotizing granuloma, consistent with tuberculosis. AFB PCR was positive for Mycobacterium tuberculosis complex. Antibiotics were altered to begin the RIPE regimen. Due to the patient's area of origin, (Micronesia is high in TB endemicity), positive IGRA, CT findings of tree-in-bud opacities with lymphadenopathy and diffuse granulomatous inflammation, and a positive PCR the patient was diagnosed with abdominal TB. The patient was discharged on long-term antitubercular therapy. | abdominal tuberculosis, antitubercular therapy, caseating granulomas, mycobacterium tuberculosis, quantiferon gold, ripe therapy | Not supported with pagination yet | null |
PMC3450927_01 | Female | 37 | A 37-year old woman, multiparous, of low socioeconomic status, was referred to our department at 23 weeks gestation for ascites. The patient reported a notion of abdominal volume increase one month ago, with night-sweat, anorexia, weight loss and abdominal pain which increases in intensity for subsequent weeks. Clinical examination revealed no fever, weight at 52Kg and height 1m60, (BMI= 20, 3), her abdomen was distended, diffused dullness on percussion, without pathological masses.
Laboratory investigation showed mild normochrom and normocytic anemia (Hemoglobin level 10,6g/dl) without leucocytosis. Abdominal ultrasound (Figure 1, Figure 2) showed intra-abdominal fluid, mainly in the lowest abdomen, and a thickened peritoneum. No ovarian mass was indentified on ultrasound. A chest X -ray showed no active lesion or old lesion compatible with pulmonary tuberculosis. A tuberculin skin test was positive. Acid-fast bacilli in sputum were not seen on sputum microscopy (Ziehl Nielson stain).
A peritoneal laparoscopic biopsy was performed to confirm the diagnosis. About 2.5 liter of straw-colored ascitis fluid was removed from the abdomen, there were multiple extensive nodules with central necrosis on the peritoneal surface and milary deposits on the intestine. Excision biopsy was taken from the peritoneal lesions. The final histologic report confirmed chronic granulomas with caseous necrosis and multinucleated giant cells. The patient received antituberculous chemotherapy regimen with rifampicin (R), isoniazid (H), pyrazinamide (Z) for two months and 4 months of RH.
The general condition of the patient improved significantly and she was discharged from the hospital on postoperative day 15. The pregnancy continued without any problem, prenatal ultrasonographic findings showed a single fetus in a cephalic presentation and biometric measurements were consistent with the date of pregnancy. The patient gave birth to a healthy infant of 3200Kg at 37th week's gestation by vaginal delivery. | peritoneal tuberculosis, diagnosis, pregnancy | Not supported with pagination yet | null |
PMC8348907_01 | Male | 53 | A 53-year-old male with a history of alcohol abuse, anxiety and insomnia presented to an outpatient clinic for his annual physical examination. He smoked half a pack of cigarettes a day for 25 years. Currently he drinks beer 2-3 times per week, but heavy liquor more frequently in the past. He denied illicit drug use and high-risk sexual activities. He was active and independent with activities of daily living. During the visit, the patient was complaining of coughing in the morning, bringing up small amounts of sputum, and of dyspnea upon exertion for some time. The patient was afebrile, and had blood pressure of 125/80 mmHg, heart rate 60 bpm. Physical exam did not have any rhonchi or wheezes. All other systems were negative. His white blood cell count was 6700/muL with 52% neutrophils, 33% lymphocytes. A chest x-ray was ordered and revealed a questionable ill-defined medial right upper lobe infiltrate, which could be related to pneumonia. However, no sputum was collected for further evaluation. There were no pleural effusions or pneumothorax. Subsequently, a computed tomography (CT) scan without contrast of the chest showed right upper and lower lobe tree-in-bud micro-nodularity in a peri-bronchovascular distribution. There were associated larger nodular opacities in the right upper lobe measuring up to 2.0 x 1.2 cm at the right apex. The patient was prescribed Levaquin for seven days, and the QuantiFERON-TB Gold test was ordered. The patient was advised to have a follow-up visit after one month. At that time, the patient felt much better and had minimal cough. However, his pulmonary function showed mild chronic obstructive defect without significant bronchodilator reversibility. The repeat CT scan of the chest showed multiple nodules in the right upper and lower lobes, especially in the azygos lobe. All these nodules were associated with a branching micronodular pattern (Fig. 1). In addition, the patient's QuantiFERON-TB Gold test was positive. The bronchoscopy was performed and showed a normal appearing endobronchial anatomy. The right lower lobe had normal findings. Attention of the scope was focused to the apex of the right lung. Multiple transbronchial lung biopsies were taken from the azygos lobe as well as the atypical subsegment of the right upper lobe. Microbiological studies revealed Mycobacterium tuberculosis in both the bronchoalveolar lavage and lung biopsy specimens. Tissue specimens were also sent for histopathology showing necrotizing granulomatous inflammation. The patient started on ethambutol, rifampin, pyrazinamide and isoniazid with vitamin B6 supplementation for two months, followed by rifampin and isoniazid with vitamin B6 for six months. Patient had been asymptomatic and no side effects so far at a follow-up visit a couple of months later. | azygos lobe, biopsy, mycobacterium, right lung, tuberculosis | A computed tomography (CT) scan of the chest showing multiple nodules, including in the azygous lobe, right upper lobe, and right lower lobe, with associated micronodular patterns. |
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