A 48-year-old woman, Jehovah’s Witness, presents to the outpatient of pulmonology service, with 8-year history of chronic cough and dyspnea, along with a progressive deterioration of her functional class (NYHA IV), along with permanent requirement of oxygen therapy and decline in the quality of life. Computed tomography scan of the chest revealed interstitial lung disease (see and ). Rheumatoid factor was positive and anticardiolipin antibodies were in indeterminate range; rest of autoimmune profile was negative. The patient did not present with clinical features of autoimmune disease. A diagnosis of autoimmune featured interstitial lung disease was made; cyclophosphamide cycle was administered without improvement of symptoms. Lung biopsy was taken via video-assisted pulmonary wedge resection, under general anesthesia. Pathology showed a histologic pattern consistent with usual interstitial pneumonia. Due to failure of medical therapy, the patient was accepted as a transplant candidate. However, due to religious beliefs, the patient stated that she does not accept blood products. Pulmonary function tests revealed a forced vital capacity (FCV) of 1.33 (44%), a forced expiratory volume in one second (FEV1) of 1.10 (44%), FEV1/FVC ratio of 83%, partial pressure of oxygen (PO2) of 91.3 mmHg and partial pressure of carbon dioxide (PCO2) of 55.1 mmHg, diffusing capacity for carbon monoxide (DLCO) 8.1 mL/mmHg/min (31%), 6-minute walk distance of 422 meters (73%), and a minimal oxygen saturation of 76 % without oxygen therapy.
Several months later, and after meticulous case review held by the lung transplant board (anesthesia, thoracic surgery, pulmonology), abdominal organ transplant board, and ethics committee, the patient was admitted for single-lung transplantation. She signed an informed consent declining transfusion of blood components, however agreeing to continuous cell-saver use. In the case of major perioperative bleeding, the patient also accepted the use of a prothrombin complex concentrate (which has advantages over fresh frozen plasma as it allows rapid correction of coagulation factor deficits with less volume infusion) and desmopressin. Blood loss during surgery was planned to be restored with 5% lactalbumin, in order to reduce the use of crystalloids in the event of significant bleeding.
The patient’s anthropometrics were 71 kg weight and 160 cm height. Regarding general procedure information, length of the procedure was 135 minutes. Cold Ischemia time was 230 minutes; there was no need for ECMO support. Bleeding during the procedure was estimated at 300 mL. Pulmonary artery pressure (PAP) was continuously measured, achieving a mean PAP of 18 mmHg in average during surgery.
Regarding hemostatic perioperative management for this patient, recommendations established in the current literature within the “Patient Blood Management” at the time were taken into account. Since preoperative hemoglobin was within normal values (14.6 g/dL), no additional measures were taken to optimize erythropoiesis. To minimize bleeding in the intraoperative setting, blood preservation methods were used: prophylactic use of antifibrinolytics was planned preoperatively, using an initial bolus of tranexamic acid 1 g, followed by continuous infusion during surgery. Fluids were adjusted at 2 to 4 cc/kg/h and readjusted according to intraoperative hemodynamic and metabolic variables. Insensible fluid loss was restored infusing a total of 870 cc of Ringer’s lactate. Normothermia was maintained with the use of Blanketrol heating system, and electrocautery scalpels were used to obtain a thorough surgical hemostasis with the help of topical hemostatic agents (tissue sealant/Surgicel). The patient agreed to intraoperative closed-system cell salvage, as it did not represent a conflict with her religious beliefs. Bleeding during surgery was calculated as of 300 cc, from which 120 cc were reinfused postoperatively. Finally, during the postoperative period, a drainage thoracostomy was positioned for a better quantification of postoperative bleeding and adequate blood evacuation from the lung. Erythropoietin was initiated during hospitalization.
After surgery, the patient was admitted to the intensive care unit, early extubation was successfully achieved, and there was no need of vasopressors or inotropes. PaO2/FiO2 ratio of 480 mmHg. Chest radiography revealed right perihilar infiltrates. The previous findings suggested a primary graft dysfunction grade 1 (see and ). Intensive care unit length of stay was 4 days, and total hospitalization length of stay was 13 days. Hemoglobin levels decreased at the end of hospitalization (see ). The patient was discharged without further complications with hemoglobin levels of 10.6 g/dL, which was treated in the outpatient setting with subcutaneous erythropoietin 2000 IU 3 times per week as ordered by internal medicine and pulmonology physicians. Hemoglobin levels increased to 11.4 g/dL at 1-week follow-up.
Pulmonary function tests after LT revealed FVC of 1.88 (53%), FEV1 of 1.73 (59%), FEV1/FVC of 92%, 6-minute walk distance of 353 meters (67%), and a minimal oxygen saturation of 94% without oxygen therapy. The patient final hemoglobin level was 11.4 g/dL.


A 41-years-old-man addicted to chronic cocaine and alcohol abuse was admitted to our emergency department in a stuporous state and with urinary incontinence. In the previous 15 days he had developed dysarthria, mutism with a prominent drowsiness and psychomotor disturbances. A brain MRI, abdomen and chest computed tomography (CT) were performed in another hospital. MRI brain scan showed multiple roundish lesions in the periventricular cerebral white matter.
On admission, laboratory tests were within normal limits, and the virological screening together with the Quantiferon, HIV and Treponemal tests was negative. Only C-reactive protein value resulted higher than normal (1.18 mg/L).
A cerebrospinal fluid (CSF) examination revealed a high albumin level, no oligoclonal bands and a normal immunoglobulin G index. The electroencephalogram (EEG) recorded low α and θ waves.
Urine toxicologic screening test detected low level of cocaine and it was negative for benzodiazepines, barbiturates, oxycodone, amphetamine, methadone and cannabinoids. The patient’s urine samples were not tested for levamisole. No myelin-oligodendrocyte-glycoprotein (MOG) or aquaporin antibodies 4 were documented.
First, the patient underwent a brain CT study which detected multiple hypointense circular lesions in the bihemispheric white matter surrounded by edema.
Brain MRI revealed multiple well-circumscribed concentric lamellar lesions in the bihemispheric white matter involving the corpus callosum,; these lesions showed alternate bands of iso-/hypointensity on T1-weighted images and alternate hypo-/hyperintensity on T2-weighted/fluid attenuation inversion recovery (FLAIR) images and presented with a rim of restricted diffusion on diffusion-weighted imaging (DWI; b = 1000), more obvious on the edge of the lesions. Apparent diffusion coefficient (ADC) map confirms alternate bands of diffusion restriction, displaying low signal intensity.
After gadolinium intravenous administration, all the lesions demonstrated an “onion-like” patchy enhancement on post-contrast T1-weighted images.
Double inversion recovery (DIR) sequences showed more detailed lamellar structure of the lesions than FLAIR images, thanks to high sensitivity in juxta and subcortical lesions detection.
MR spectroscopy revealed elevated choline and lactate peaks and decreased N-acetylaspartate peak ().
MRI of the spine showed no evidence of demyelinating lesions. Stereotactic biopsy obtained from frontal right paraventricular lesion showed gliosis and many swollen reactive eosinophilic astrocytes with perivascular cuffing. Axonal stain showed long stretches of intact axon. In addition, perivascular and periventricular macrophages were found (). These pathologic findings supported a possible inflammatory/immune-mediated etiology.
The patient was treated with high-dose oral methylprednisolone for 15 consecutive days, after which his clinical symptoms partially improved.


A 34-year-old male patient with Norwood Stage 5 hair loss was wearing skinhead hair style with hair length less than 1 mm (Fig. ).


A 40-year-old male patient with advanced alopecia of Norwood stage 6 had been wearing closely shaved hair style for more than a decade (Fig. ).


A 43-year-old male patient who had received hair transplant surgery by FUT method twice and by FUE once with dissatisfying results underwent shaved hair style SMT with a concomitant procedure at FUT his donor scar (Fig. ).


We report on an 80-year-old male patient who suffered a cryptogenic ischemic stroke in the right cerebellum in April 2012. The patient was included in the longitudinal Determinants of Dementia after Stroke (DEDEMAS) study [] and underwent serial standardized magnetic resonance imaging (MRI) in combination with 18F-florbetaben [] Aβ positron emission tomography (PET) at 6 months according to the study protocol (NCT01334749). Amyloid PET at baseline demonstrated mild positivity. At the follow-up visit in 2015 the patient demonstrated an incident mild cognitive impairment diagnosed by Petersen criteria [] and an incident focal cSS.A diagnostic lumbar puncture revealed reduced Aβ levels (Aβ42 = 279 pg/mL), a reduced Aβ42/40 ratio (3.8%), and a normal total tau level (378 pg/mL) in CSF.
The follow-up MRI in 2017 showed a progression of cSS from focal to disseminated and twelve incident cerebral microbleeds restricted to lobar regions in the right hemisphere. Hence, a diagnosis of probable CAA according to modified Boston criteria [] was made. All cSS-affected sulci were segmented using ITK-Snap for analysis as previously described []. The patient further underwent a follow-up Aβ PET and 18F-PI2620 tau PET [] in 2017/2018.
Follow-up Aβ PET 6 years from baseline demonstrated a tremendous increase of fibrillar amyloid deposition predominantly in the contralateral left hemisphere not affected by cSS (Fig. ). In contrast, tau deposition was predominantly localized to the right hemisphere partly overlapping with brain regions affected by cSS. This was also reflected by plotting asymmetry of tau PET against Aβ PET (Fig. ) demonstrating an opposite dominance of amyloid and tau deposition. We further performed a segmental analysis of single brain regions affected by cSS (Fig. ) and found that amyloid deposition was not different in the vicinity of cSS compared to other compartments (Fig. ). In contrast, tau depositions were elevated in the vicinity of cSS-affected regions compared to non-cSS-affected brain regions (Fig. ).


A 56-year-old woman, with a known HH C282Y mutation and on phlebotomy when required, moved to Qatar 2 years ago. She was referred to the hematology clinic for further evaluation and to continue the medical care, as well as for an evaluation whether she still required further phlebotomy.
On her initial visit, she seemed to be in a fair general condition, and her clinical examination was not remarkable. Her complete blood count showed a white blood cell count of 6.8 × 103/μl (normal range 4–10), Hb of 14.5 g/dL (normal range 12–15), platelets of 208 × 103/μl (normal range 150–400), serum ferritin of 287 μg/L (normal range 18–340). An MRI of the liver was done which showed no evidence of iron overload: 0.9 mg/g dry tissue (normal range 0.17–1.8), 16 mmol/kg dry tissue (normal range 3–33). She was again evaluated 2 years later, when her serum ferritin level was found to be at 482 μg/L (normal range 18–340). Therefore it was decided to do a heart and liver MRI which showed <1.2 mg iron/g dry weight (no siderosis) in the heart and 3.4 mg iron/g dry weight (no siderosis) in the liver. There was no iron overload neither in the heart nor in the liver, which demonstrates the superiority of T2* MRI over serum ferritin.


A 76-year-old man was diagnosed with squamous cell carcinoma of the lung (cT2aN2M0, c-stage IIIA). He regularly took edoxaban for atrial fibrillation, and he received chemoradiotherapy (2 Gy × 33 fractions with weekly carboplatin and weekly paclitaxel). Nine days after the end of thoracic radiation, he developed a fever of 38.6°C. Chest computed tomography (CT) showed a new GGO in the irradiation area. We suspected radiation pneumonitis and then initiated oral prednisolone therapy (initial dose: 1 mg/kg/day). We gradually reduced the dose and then discontinued prednisolone for 6 months. When the dose of prednisolone had decreased to 10 mg/day, 41 days after the thoracic radiotherapy, we initiated durvalumab maintenance treatment (10 mg/kg, bi-weekly).
After 11 cycles of durvalumab therapy, 3 weeks after the discontinuation of prednisolone, he developed dyspnea and malaise. His body temperature was 37.3°C, heart rate was 75 beats per minute, blood pressure was 111/81 mm Hg, and oxygen saturation on room air was 96%. Chest CT showed new GGOs in the bilateral lower lobes, extending beyond the irradiated areas (Fig. ). Laboratory blood tests exhibited an increase in C-reactive protein (8.24 mg/dL) and KL-6 (515 U/mL). He was hospitalized and underwent fiberoptic bronchoscopy on hospital day 2. The BALF from the right B8 gradually became bloody (Fig. ), and revealed a cell count of 13.4 × 105 cells/mL with 70% neutrophils, 10.5% lymphocytes, 11% eosinophils, and 8.5% macrophages. Despite immediate discontinuation of edoxaban and administration of ciprofloxacin, his symptoms and respiratory conditions rapidly deteriorated. Thereafter, although there was a negative bacterial culture in the BALF, we detected hemosiderin-laden macrophages (Fig. ). Myeloperoxidase ANCA (antineutrophil cytoplasmic antibody), proteinase-3 ANCA, and anti-glomerular basement membrane antibody were all negative. Thus, we suspected durvalumab-induced DAH. On hospital day 7, we initiated oral prednisolone (1 mg/kg/day). Chest CT on hospital day 10 showed an expansion of GGOs to the left lung (Fig. ). A methylprednisolone pulse (1,000 mg/day) was administered for 3 consecutive days, followed by oral prednisolone (1 mg/kg/day). On hospital day 14, due to his deteriorated respiratory condition, we introduced invasive mechanical ventilation with endotracheal intubation. On hospital day 15, we administered cyclophosphamide (750 mg/m2). The second BALF from the left B5 and B9 on hospital day 24 were both bloody and contained hemosiderin-laden macrophages. Due to exacerbated respiratory failure, the patient died on day 50.
We performed an autopsy 14 h after his death. In the macroscopically bloody lung areas with decreased aeration (Fig. ), the alveolar spaces were microscopically filled with red blood cells, which suggested fresh bleeding (Fig. ). In the macroscopically white lung areas, hemosiderin-laden macrophages were still present, which suggested old bleeding (Fig. ). Almost all parts of the lungs exhibited one of these two histological findings. Neither pulmonary vasculitis nor diffuse alveolar damage was detected in the autopsy. Immunohistochemical analyses showed that CD3-positive (CD3+) and CD8+ lymphocytes were present in the lung interstitium, but not around capillaries. On the other hand, CD20+ and CD4+ lymphocytes were scarcely observed (Fig. ).


A 73-year-old Caucasian male with a family history of cancer (his father and brother had prostate cancer and his sister had breast cancer) presented to his family physician with a 6-month history of a mass located medially on his right upper thigh. There was no history of trauma, the mass was nontender, and it had not grown in size. There were no systemic symptoms including fever chills, night sweating, or unintentional weight loss. He denied any nausea, vomiting, or generalized malaise. He had no prior history of smoking or tobacco product use, denied the use of alcohol, and had no history of being immunocompromised.
Physical examination revealed a subcutaneous firm, nonmobile, not warm, irregular mass of approximately 4 cm on the patient's right upper outer thigh with no associated inguinal lymphadenopathy. The patient's abdomen was soft, nontender, with no masses, hepatomegaly, or splenomegaly. The patient's afflicted leg showed no signs of muscle weakness, and the range of motion was normal.
A complete blood count (CBC) demonstrated a WBC of 7.9 K/μL (normal 4.8–10.8) with a normal differential, Hb 5.84 M/μL (normal 4.70–6.10), Ht 50.7% (normal 42.0–52.0), and platelet count 243 K/μL (normal 164–446). His electrolytes, liver and renal functions were normal. An ultrasound of the right leg was performed and showed a vascularized heterogeneous mass measuring 5.9 × 3.8 × 1.3 cm within the subcutaneous soft tissues, consistent with either a primary muscular or metastatic tumor.
A subsequent magnetic resonance imaging (MRI) with contrast of the right lower extremity (Fig. ) demonstrated a 5.4 × 3.4 × 1.5 cm mass which was noted to be T2 hyperintense, T1 slightly hyperintense to muscle, and which avidly enhanced in a uniform fashion following contrast administration. The mass was found to be superficial to the rectus femoris muscle with no signs of muscular invasion. The remainder of the thigh was normal on imaging. Several small bilateral inguinal lymph nodes were noted, all within normal limits. At this point, the imaging findings were still primarily concerning for a soft tissue sarcoma or metastasis.
A contrast-enhanced computed tomography (CT) of the chest, abdomen, and pelvis revealed bilateral axillary lymphadenopathy. The liver, pancreas, adrenal glands, and kidneys were unremarkable. Multiple enlarged gastro-hepatic lymph nodes, the largest measuring 5 × 3 cm in size, were visualized. Multiple mildly prominent retroperitoneal lymph nodes were also present, the largest measuring up to 10 mm in the short axis dimension. Based on these findings, the differential diagnosis included metastatic adenopathy and lymphoma.
Microscopic examination of a right thigh biopsy demonstrated sheets of intermediate-sized lymphocytes with somewhat dispersed chromatin, occasional inconspicuous nucleoli, irregular nuclear contours, and scant amphophilic cytoplasm (Fig. ). Brisk mitotic activity was noted. The neoplastic cells were strongly and diffusely positive for CD20, CD5, and cyclin D1, positive for BCL2, and negative for CD3, CD10, CD23, and BCL6. The proliferation index was estimated at >95% based upon Ki-67 expression. A subsequent biopsy of an enlarged celiac lymph node showed similar morphology and demonstrated kappa-restricted B cells strongly positive for CD20 and CD5 by flow cytometry. These results were consistent with MCL. TP53 sequencing was not performed.
A post-biopsy fluorodeoxyglucose-positron emission tomography (FDG-PET) scan (Fig. ) revealed abnormal uptake within lymphadenopathy in the neck, bilateral axilla, mesenteric, retroperitoneal, and bilateral pelvic regions. There was also diffuse uptake in the spleen and abnormal uptake in the known right anterior thigh lesion. This is consistent with MCL involvement.
A bone marrow aspirate demonstrated abundant bone marrow particles for review. Numerous megakaryocytes were noted, most demonstrating normal lobulated nuclei. There was trilineage hematopoiesis with maturation and progression of the myeloid series through the segmented neutrophil. No significant dysgranulopoiesis was seen. There was no morphologic increase in blast cells. Numerous erythroid precursors were noted demonstrating normoblastic maturation. No significant dyserythropoiesis was seen. Scattered small- to intermediate-sized lymphocytes were noted and did not appear to be increased in quantity. Occasional plasma cells were seen, not increased in quantity. Flow cytometry showed 2 small monoclonal B-cell populations, one with CD5-positive B cells (2% of lymphoid cells) and a separate one with CD10-positive B cells (5% of lymphoid cells) both showing kappa light chain restriction. CD34+ cells (2.4% of total cells) were not increased. In conclusion, the bone marrow aspirate presented no morphologic or immunohistochemical evidence for bone marrow involvement by MCL. A bone marrow biopsy demonstrated cellularity averaging approximately 50% with areas of intramedullary hemorrhage. Scattered megakaryocytes were adequate in quantity with most appearing to demonstrate normal lobulated nuclei. Trilineage hematopoiesis with maturation was present. There appears to be a mild erythroid hyperplasia. The myeloid to erythroid ratio appears to be decreased and was estimated at approximately 1:1. There was no obvious increase in blast cells. There was no obvious increase in plasma cells. No lymphoid aggregates or atypical lymphocytes were seen. No morphologic evidence for bone marrow involvement by MCL was seen.
The patient went to UC Davis' Comprehensive Cancer Center for therapy that consisted of R-CHOP (rituximab plus cyclophosphamide, doxorubicin, vincristine, and prednisone) alternating with R-DHAOx (rituximab plus dexamethasone, high-dose cytarabine, and oxaliplatin), a slight modification of standard R-CHOP alternating with R-DHAP (rituximab plus dexamethasone, high-dose cytarabine, and cisplatin) as cisplatin was substituted with oxaliplatin due to the patient's age []. After 6 courses of chemotherapy, a PET-CT showed complete full metabolic remission. He proceeded to receive high-dose chemotherapy consisting of R-BEAM (Rituxan, BCNU, etoposide, cytosine arabinoside, and melphalan) followed by autologous stem cell transplantation/rescue as consolidation. At present, he is alive with no evidence of disease 12 months following diagnosis.


A 60-year-old male with the chief complaints of progressive abdominal pain and poor appetite was admitted to our department.
He previously underwent surgery for a splenic CE lesion. There was no other relevant personal or familial history.
Physical examination upon admission revealed a mass in the left upper quadrant of the abdomen.
Laboratory examinations (routine blood tests, routine urine tests and urinary sediment examination, routine fecal tests and occult blood test, blood biochemistry, immune indices, and infection indices) showed no apparent abnormalities.
Imaging examination with computed tomography and angiography revealed two irregular CE2 lesions (approximately 10.0 cm × 14.0 cm × 13.6 cm and 12.5 cm × 11.2 cm × 5.8 cm), respectively, located in the left upper retroperitoneal cavity and the abdomen, according to World Health Organization (WHO) classification (Figure and )[,]. The lesions mainly invaded the liver-spleen-gastric gap, superior section of the spleen, left retro-renal cavity and the abdominal aortic wall (Figure and ). There was no other abdominal organ involvement.


A 52-year-old woman presented with intermittent left lower quadrant pain that had started in 2017. She was diagnosed with systemic lupus erythematosus 19 years ago and had been taking oral corticosteroids since then. Her past medical history also showed steroid-induced diabetes mellitus, hypertension, and hypothyroidism, which was treated with levothyroxine. Menopause occurred when she was in her thirties.
On her first visit to the emergency department in another tertiary hospital in May 2018, she underwent abdominal pelvic computerized tomography (APCT), which showed a multiloculated lesion on the left adnexa, with left ovary involvement and hydrosalpinx or pyosalpinx. Under the impression of a TOA, she was admitted and received conservative care including intravenous (IV) antibiotics for 15 days. However, the pain in her left lower quadrant persisted. She visited the emergency department for the second time in July 2018. APCT was performed again, which revealed a slightly increased multiloculated, rim-enhancing lesion compared with the previous APCT findings. Again, the impression was TOA; however, APCT showed a newly developed wall enhancement of the left ureter due to either secondary change or combined ureteritis. Because of persistent pain, she agreed to undergo surgery. On August 3, 2018, the patient underwent laparoscope-guided bilateral salpingo-oophorectomy. The pathology report showed chronic active inflammation with foam cell collection in her left ovary and both salpinges.
Because the surgery did not alleviate her pain, she underwent both APCT and magnetic resonance imaging in April 2019, which revealed a 7-cm irregular mass in her left adnexa and an infiltrative lesion in her uterus, suggestive of endometrial cancer. Imaging results also showed left ureter invasion by the left adnexal tumor. As prophylactic management, a left ureter stent was inserted. Furthermore, she underwent dilatation and curettage for diagnosis. Pathological examination showed chronic active inflammation with foam cell collection, consistent with the finding in the previous laparoscopic surgery.
The patient visited the emergency department of our hospital on May 20, 2019. She had sepsis and a body temperature of >39°C; blood culture revealed the presence of extended-spectrum beta-lactamase–producing Escherichia coli. The cervix culture taken at the emergency department also showed the presence of the same organism. Her initial blood urea nitrogen level was 45.6 mg/dL, and her creatinine level was 2.75 mg/dL, which implied septic postrenal acute kidney injury. For septic shock evaluation, she underwent APCT without contrast, which revealed newly developed mild hydronephrosis of the left kidney. The left adnexal mass remained the same as that seen in April 2019 (). We started initial shock management in the emergency department and conservative care including an IV antibiotic (meropenem) for bacteremia and left percutaneous nephrostomy (PCN) for left hydronephrosis and acute kidney injury.
After her recovery from septic shock, the patient underwent debulking surgery on June 4, 2019. During the surgery, severe adhesion in the pelvis involving the peritoneum, rectum, uterus, and left ureter was noted. Multiple hard and catheter-shaped foreign body–like materials were observed in the left parametrium and left pararectal space and near the left internal iliac artery. Caseous necrotic materials had spread deep into the left pelvic and paravaginal space. Total hysterectomy and debulking operation of the left pelvic mass, including left pelvic lymph nodes, were performed.
The final pathology showed foamy histiocyte aggregation and acute and chronic inflammation with numerous Michaelis-Gutmann bodies, consistent with malakoplakia (). According to the recommended treatment for malakoplakia, we continued IV administration of tazoperan after the surgery. After the culture from peritoneal fluid cytology showed ceftazidime-sensitive E. coli, we changed the antibiotic to ceftazidime 1 g twice a day.
However, her creatinine level increased gradually to 4.48 mg/dL after the surgery. A retrograde pyelogram showed patent left ureter and dye spillage from the right ureter. Right PCN was performed. A computerized tomography scan performed on postoperative day 9 showed recurrence of a 9-cm mass in her left pelvis. Hence, we again administered tazoperan. On postoperative day 21, the patient was referred to a general hospital for further conservative care with IV antibiotics.
On July 24, approximately 1.5 months after surgery, she underwent left PCN removal and right PCN clamping. On August 2, the patient returned to the emergency department with complaints of fever and right flank pain that had started on the previous day. She presented with right costovertebral angle tenderness and pyuria and was admitted to the urology department with a diagnosis of right acute pyelonephritis. On August 9, the anterograde pyelogram showed leakage at the right ureter, which was similar to the previous retrograde pyelogram finding. Right PCN exchange was performed. After conservative care with IV antibiotics, the patient was discharged on August 12. On September 10, direct ureteroneocystostomy was attempted; however, only right PCN exchange was performed because the ureter was short.
The patient has been closely followed up through outpatient visits to both urology and obstetrics and gynecology departments.


A 49-year-old woman presented with a 5-month history of painful swelling and occasional pus discharge from the tip of the right great toe. To alleviate the pain and swelling, she had at once blindly clipped her nail at the distal end, underneath the swollen nail fold. Examination revealed a tender enlarged lateral nail fold with the nail plate at the medial end showing a pointed spur at the distolateral end. There was dimpling at the tip corresponding to the direction of the spur []. No active discharge or granulation tissue was observed. A provisional diagnosis of ingrowing toenail (Heifetz stage 1) was considered.
She was surgically managed with lateral plate avulsion followed by chemical cauterization of the lateral horn of the matrix with 10% sodium hydroxide for 1 min. The fistulous tract was deroofed and completely excised [Figure and ]. The wound healed well following surgery. Follow-up at 4 weeks after surgery was uneventful with complete epithelialization of the wound [].


A 56-year-old female without personal and familiar history of tumors presented to our department on September 2018 with a right-sided painless cervical swelling. Tumor onset was referred to be 7 months before she came to our attention. The mass presented with a progressively enlargement. Neither history of trauma nor recent upper respiratory tract infections were referred. Compressive symptoms were not also referred.
On physical examination, the mass was located in the right submandibular region. Skin overlying the mass was not compromised. It was soft to tender in consistency, lobular with ill-defined margins, painless and freely movable, and cervical lymphadenopathy was not clinically detected (). Normal function of the facial nerve was observed. No alteration of the lingual nerve sensitivity was referred.
Ultrasonography (US) revealed a cervical anechoic multilobulated mass in the submandibular space.
Magnetic resonance imaging (MRI) was performed and showed a cystic mass of 4 × 4 × 3.5 cm in size located under the right mandible and strictly adherent to the submandibular gland. On T2-weighted images, the mass resulted hyperintense, while hypointense in those T1 weighted ().
A branchial cleft cyst was firstly supposed.
Surgery was performed under general anesthesia. Horizontal incision was made about two fingers below the mandible in order to avoid marginalis mandibulae nerve injury. The mass was cleaved from the submandibular gland and completely removed, and then the surgical specimen was sent for histopathological examination.
Macroscopically, cysts bigger than 2 cm3 were observed. Microscopically, the mass was composed by a variety of dilated lymph vessels involved in a fibrovascular stroma (). Diagnose of completely excised CL was done.
No facial nerve injury was noticed after surgery.
A follow-up was programmed. At the date, after 18 months from surgery, no relapses were noticed.


Patient A, a 48-year-old man, visited the spine center at the University Hospital with pain in the right buttock, lateral thigh, and calf, which had lasted for two weeks. The pain was piercing and tingling. The patient had a score of 7 out of 10 on the numeric rating scale (NRS). The pain was aggravated when he flexed his lower back. Upon physical examination, he had a positive straight leg-raising test (SLRT) result at 40 degrees on the right side. His right extensor hallucis longus had a Medical Research Council (MRC) score of 3, but no sensory deficits were observed. Lumbar magnetic resonance imaging (MRI) revealed a massive right central extrusion of the L4-5 intervertebral disc (). Before he visited our hospital, he had received three repeated transforaminal ESIs at the right L5 nerve root, as well as two repeated interlaminar ESIs at L4-5. However, no pain reduction was observed following each procedure. At his next visit to our hospital, we conducted a percutaneous disc decompression with radiofrequency energy using the navigable percutaneous disc decompression device (L’DISQ®, U & I Co., Uijeongbu, South Korea) (). The procedure was conducted under the guidance of fluoroscopy. The patient was in the prone position, and we marked the patient’s skin approximately 13 cm from the midline in order to guide the needle entry. The endplates of the target disc (L4-5) space were aligned. The C-arm was rotated ipsilaterally in order to position the lateral margin of the ipsilateral superior articular process. This was done at approximately 3/5 of the distance across the vertebral body, as visualized in the oblique position (typically rotated 15 degrees from a zero-degree lateral projection). The C-arm was rotated into the contralateral side (left) of the disc herniation. After skin sterilization and anesthesia, we inserted a 16-gauge introducer needle, directing it gently toward the annular surface of the contralateral foramen (slightly lateral portion of the lateral edge of the superior articular process) using fluoroscopy. Once the disc surface was touched, the introducer needle was advanced approximately 1 cm into the disc. After confirming the introducer needle position using anteroposterior (AP) and lateral views, the stylet was removed, and the wand was inserted through the introducer needle. The wand had a navigable tip that could be curved to the desired angles by rotating a control wheel on the handle. We introduced the wand, and it passed the midline of the targeted disc. Using the control wheel, we placed the tip of the wand into the center of the posterolateral portion of the herniated disc. Before the radiofrequency current was applied, motor nerve stimulation was used to confirm that the tip of the wand was not close to the nerve root. Then, the radiofrequency current (265 Vrms) was applied to the herniated portion of the disc using an RF generator (L’DISQ®, U & I Co., Uijeongbu, South Korea). During the decompression, the tip of the wand was continuously moved back and forth to increase the ablated disc volume. The 5-second ablation was performed 60 times. The procedural duration was 30 minutes. After the procedure, the patient reported no adverse effects. One week after the decompression procedure, the patient’s pain score had reduced from 7 to 2. At the 1-, 3-, and 6-month follow-ups, the patient’s NRS pain score was 1. At his 2-year follow-up after the procedure, he reported that his radicular pain was sustained at an NRS score of 1, and no oral medications or procedures were required.


Patient B was a 39-year-old man who visited the spine center at the University Hospital with pain in the right buttock, posterior thigh, and calf, which had lasted for 15 days. His NRS score was 8 out of 10, and he could neither sit nor stand due to severe pain. The pain was piercing and tingling. Upon physical examination, he had a positive SLRT result at 30 degrees on the right side. His MRC score for the right extensor hallucis longus and plantar flexor was 4. Hyperalgesia and hyperesthesia were observed at the right S1 dermatome. On lumbar MRI, a large right central intervertebral disc extrusion was observed at the L5-S1 intervertebral disc (). Before his visit to our hospital, the patient had received 3 transforaminal and 2 interlaminar ESIs. However, no pain reduction was observed after each injection. We performed a percutaneous disc decompression under the guidance of fluoroscopy using the device and techniques that were used for patient A (). Post-procedural adverse effects were not observed. One week after the procedure, the patient’s pain had reduced with a change in the NRS score from 8 to 1. At the 1-, 3-, and 6-month follow-ups, as well as the 2-year follow-up, the patient reported that his pain had completely resolved. Additionally, his motor deficits were nearly completely recovered.


A 16-year-old boy developed bilateral painful perniotic-like plaques on his toes (Figure ). He had been confined to his home and denied any significant cold exposure. His brother had a cough and fever several weeks earlier, which resolved without viral diagnostic testing. A nasopharyngeal swab was negative for SARS-CoV-2. Serologies to assess for past SARS-CoV-2 infection were not performed.


A 48-year-old female resident of New City, New York, developed fever, mild leucocytosis, a purpuric macular skin rash of the legs and perniotic lesions on the toes. A skin biopsy was performed on one of the toe lesions. Her nasopharyngeal swab test for COVID-19 was negative. Her COVID-19-associated IgG and IgM serologies were also negative.


The patient was a 65-year-old woman who lived in very close proximity to the South Dakota Smithfield Pork processing plant, which had experienced a COVID-19 outbreak affecting 700 of its 3000 employees. At the time of the outbreak she developed distinct papular lesions of the fingers accompanied by a cough. Over the ensuing weeks the lesions underwent full resolution. She declined COVID-19 testing.


A 52-year-old man was referred to our dermatology department with painful, coalescent, erythematous, papulo-infiltrative, nodular plaques on his midface. He has a history of uncontrolled diabetes mellitus Type II and a history of CL that started 3 years ago as a slowly growing papule on his nose that evolved to a nodule. Direct smear was positive for leishmaniasis bodies. The patient was treated with systemic meglumine antimoniate (glucantime) for 20 days; the lesion was healed with a scar. Two years later, the patient had a recurrence of the lesion at the border of the scar; he sought folk medicine that resulted in severe ulcers and dissemination of the lesion, which led to deformity of the nose. Results of dermatologic examination revealed extensive erythematous papules and nodular plaques on his nose, upper lip and cheeks with telangiectasia on his cheeks and severe ulcers and scarring on the nose and upper lip (). The other parts of his face were spared. There was no evidence of systemic involvement. The results of the routine lab tests, including complete blood count (CBC) and serum biochemistry are listed in (). Smear was not made since the patient is known to have a history of leishmaniasis. Histological examination showed changes in the dermis with epidermal atrophy. There was a mixed inflammatory infiltrate composed of lymphocytes, histiocytes, plasma cells and multinucleated cells of Langhans (). Neither parasites within dermal macrophages (), nor tuberculoid granulomas with caseation necrosis were detectable. Diagnosis of LCL was established based on the clinical aspect, facial localization, chronic evolution, the history of CL and histopathologic findings even with the absence of intra-amastigote in the biopsy. Patient was admitted in the hospital and was started on systemic meglumine antimoniate (60 mg/kg/day) divided into two doses for 20 days in addition to ceftriaxone (500 mg twice a day) with application of topical cream diprogenta (betamethasone dipropionate and gentamicin sulfate). During the treatment, the patient was monitored daily for any clinical or laboratory signs of chemical pancreatitis, liver function tests, CBC in addition to a daily electrocardiography (ECG) test. On Day 10, the ECG showed a slight prolongation of QTc (QTc = 459 msec) for only 1 day. No other significant side effects were observed. At the end of the Day 20, improvement was noted and the patient was discharged and started on hydroxychloroquine (plaquenil) (200 mg twice a day) for a month. At the end of the month, he came for a follow-up and the lesion was completely healed ().


A 12-year-old female accompanied by her father reported to the Department of Pedodontics and Preventive Dentistry, Dental Institute, Rajendra Institute of Medical Sciences, Ranchi, Jharkhand, India, with the chief complaint of enlarged gums in the upper and lower jaws along with difficulty in eating and speech and was unable to close her mouth. Growth was progressive from the age of 3 years. The patient had stopped going to school from the last 2–3 years because of ghastly appearance and had minimum social interaction. Familial, medical, and drug histories were noncontributory. Extraoral examination showed an exaggerated convex profile, a highly incompetent compressed lip, and a tissue bulge protruding out of the mouth, causing partial obstruction of the nostrils and partial obliteration of the nasolabial fold bilaterally. Intraoral examination showed massive generalized Grade III enlargement of the gingiva, which almost covered the crowns of all teeth, with partial cusp visibility of 26, 75, 84, and 46 []. Color of the enlarged gingiva was pink with melanin pigmentation. The enlarged gingiva was firm and fibrous in consistency. The excessive growth had caused obliteration of the vestibular fold []. The girl was chewing food with the assistance of the enlarged gums. Orthopantomogram showed multiple retained deciduous teeth namely 55, 54, 65, 75, 74, 85, 84, and 83. It also showed that all permanent teeth were present with displaced permanent molars; however, no significant bone loss was found []. Computed tomography scan showed protruded maxillary and mandibular anterior alveolar segments along with soft-tissue overgrowth with proclination of teeth, suggesting migration of teeth as well as expansion of the alveolar bone secondary to fibrous growth [].
Complete hemogram including hormonal profile consisting of thyroid test, parathyroid test, calcium, and alkaline phosphatase was done. All the tests were within the physiological limits.
A small tissue measuring 0.5 cm × 0.4 cm from the lower jaw was sent for biopsy, which showed proliferation of the fibrocollagenous tissue. No evidence of malignancy was found. Based on history and clinical features, a provisional diagnosis of IGF was made. Because of such massive gingival enlargement and considering patient compliance, excision of the enlarged gingival mass by gingivectomies along with anterior segmental ostectomy under general anesthesia was planned.
Among the suggested treatment protocols, surgery was done with the help of electrocautery using ledge-and-wedge technique along with internal bevel gingivectomy. All the retained deciduous teeth along with the permanent anterior teeth were removed as they were significantly mobile []. The excised mass was sent for histopathological analysis, which confirmed the findings of previous histopathological report. Hematoxylin- and eosin-stained section showed stratified squamous epithelium with rete pegs overlying fibrous connective tissue stroma. Higher magnification showed connective tissue comprising of uniform spindle cells with abundant intercellular matrix []. The child was kept on observation and follow-up, which gradually showed improved lip competency and esthetics in the next few weeks. Two months later, a temporary prosthesis was made for esthetic improvement []. The girl and her parents were motivated to start her schooling and normal day-to-day activities. There was a significant gain in child's confidence after removable prosthesis. Permanent implant-supported prosthesis is planned at a later stage after completion of the growth of jaw.


A 10-year-old girl presented to our hospital with a 1-month history of a progressively swelling mass and limited flexion of her left index finger. She had no apparent trauma history. A swollen and painless immovable mass was observed in the proximal interphalangeal (PIP) joint of the affected finger, without skin ulcers or redness (). A physical examination revealed that the active flexion of the PIP joint angle had decreased to 85°, while her flexor strength was normal. The patient’s other joints and fingers were not affected. Her parents denied a history of tuberculosis, rheumatoid arthritis, or any other inflammatory diseases. Laboratory studies showed that the white blood cell count, high-sensitivity C-reactive protein level, erythrocyte sedimentation rate, and tumor marker levels were all within normal limits. The rheumatoid factor, anti-cyclic citrullinated peptide antibody, anti-streptococcal hemolysin O, and tuberculous (tuberculosis antibody and T-SPOT.TB) test results were all negative. Radiographs showed swelling of the soft tissue without bone involvement in the PIP joint (). A chest radiograph was normal without hilar lymphadenopathy or tuberculomas. Ultrasound showed good continuity of the flexor digitorum profundus (FDP) tendon, with hyperechoic lesions both inside and outside it. The lesion moved with the tendon. Magnetic resonance imaging (MRI) demonstrated a well-defined lesion isointense to muscle on T1 sequences and hyperintense to muscle on T2 sequences, and strong homogeneous enhancement was observed after injection of contrast medium during coronal plane sequencing. The enhanced-signal lesion surrounded and partly infiltrated the FDP tendon, manifesting as a double ring sign on transverse plane sequencing ().
The mass was misdiagnosed as a tenosynovial giant cell tumor (TGCT) when she initially visited a doctor at a community hospital. MRI was performed when she presented to our department, revealing a lesion within the tendon, which was an uncharacteristic finding. Conservative treatments were attempted when she first presented to the hospital, but she developed a limitation of finger flexion within a short time. Her parents consented to surgical treatment. During the operation, a volar Brunner zig-zag incision was made, and the subcutaneous lesion was exposed. The A2, A3, and A4 pulleys were all intact. During the operation, the entire A3 pulley and distal two-thirds of the A2 pulley were removed, and the proximal part of A4 was slightly cut open. The FDP tendon was found to be expanded. Hypertrophic synovium surrounded the outside of the tendon (). The synovium invaded the tendon sheath, which could not be separated. After removal of the peritendinous tissue, the flexor tendon was confirmed to be intact. It was then dissected longitudinally. A large amount of synovial-like substance was found within the tendon (). This substance was completely cleared, and the tendon was then repaired with an uninterrupted suture (). The skin wound was closed. Pathologic examination revealed synovitis with synoviocytes and inflammatory cell infiltration, without hemosiderin deposition or multinucleate giant cells ().
The patient received cefuroxime (Glaxo Wellcome Operations, Hertfordshire, UK) at 0.25 g twice a day for 3 days after the surgery. Active finger motion was promoted by physical therapists at the end of the first week. The sutures were removed at the end of the second week. A brace with the index finger in a functional position was used for 3 weeks. A physiotherapist supervised the patient’s rehabilitation until full active range of motion was achieved. Normal tendon gliding returned without recurrence by the 1-year follow-up. The active flexion angle of the PIP joint improved to 100°.


A 78-year-old man with high-grade T1 urothelial carcinoma status post-transurethral resection of bladder tumor who had been on once-weekly intravesical BCG instillation for 2 months presented to the emergency room (ER) with fever, rigors and altered mental status.111111 He also reported shortness of breath but denied cough or chest pain. He had no urinary or gastrointestinal symptoms. He had his most recent BCG instillation done the day before the index presentation. Vital signs revealed a temperature of 102.8 F, pulse rate of 92/min, respiratory rate of 40/min and blood pressure 78/45 mmHg with oxygen saturation of 93% on ambient air. On physical examination, he appeared toxic, dehydrated and in respiratory distress. Chest examination revealed asymmetric chest expansion with dullness to percussion, decreased tactile fremitus on the left side with reduced air entry on auscultation. The cardiovascular examination was however unremarkable.
Complete blood count revealed marked leukocytosis with a white cell count of 27,200/microliter (Normal 4,800–12,000/microliter) and hemoglobin of 11.5 g/dL (Normal 14.0–17.5 g/dL). Lactate was markedly elevated at 7.8 meq/L (Normal 0.6–1.4 meq/L) and arterial blood gas revealed high anion gap metabolic acidosis. Basic metabolic panel revealed creatinine of 2.31 mg/dL from normal baseline (Normal 0.6–1.3 mg/dL) and blood urea nitrogen of 28 mg/dL (Normal 7–25 mg/dL). Sputum culture with gram stain showed normal oral flora. Urinary streptococcal and legionella antigens were negative. Chest X-ray on admission revealed left basilar pneumonia with small left pleural effusion. CT head showed no acute intracranial abnormalities. The preliminary blood culture result was negative for any micro-organisms.
Due to altered mental status with increased work of breathing and acute hypoxemic respiratory failure, patient was intubated and placed on ventilation in the medical intensive care unit. He was commenced on vasopressors and intravenous Vancomycin and Piperacillin-Tazobactam for septic shock thought to be secondary to community-acquired pneumonia and possible urinary tract infection. Despite the completion of several courses of broad-spectrum antibiotics, he continued to have low-grade pyrexia and failed to improve clinically. Suspicion for rarer etiologies such as systemic BCG dissemination began to grow and blood cultures for acid-fast bacilli initially obtained on admission returned positive for acid-fast bacilli, specifically Mycobacteria bovis in the two sets of samples thus confirming our suspicion. Broad-spectrum antibiotics were immediately discontinued, and he was commenced on triple therapy with Isoniazid 300 mg daily, Ethambutol 1600 mg daily and Rifampicin 30 mg daily. He improved clinically and was discharged 5 days later to complete 2 months of triple therapy with Isoniazid, Rifampicin and Ethambutol followed by 4 months of maintenance therapy with Rifampicin and Isoniazid. He was planned to return for follow up with the infectious disease team.


A 54 year old woman with a past medical history significant for Roux-en-Y procedure
11 years ago and a recent exploratory laparotomy for small bowel obstruction 3 months ago,
with no prior history of IV drug abuse, presented to the emergency department with altered
mental status described as confusion and hallucinations, and chest pain over the past
3 days. Vitals at initial assessment were: blood pressure (BP): 119/78 mmHg, pulse 85 beats
per minute, temperature 97.8 F°, respiratory rate (RR): 18/minute and SpO2 of 84%
at room air. On examination, patient had 13/15 on Glasgow Coma Scale (GCS), patient was
alert and awake but not oriented to person, place or time, lung exam revealed bilateral
coarse crackles and expiratory wheezes throughout lung fields, and cardiovascular exam
revealed regular rate and rhythm, normal S1 and S2, no murmurs or gallops. Electrocardiogram
(ECG) showed normal sinus rhythm with no ST or T wave changes. Abdomen examination showed
present bowel sounds with mild diffuse tenderness to palpation and a 3 cm abdominal wound
healing well with no surrounding erythema or exudate from recent exploratory laparotomy.
Initial laboratory results showed a white blood cell count of
11.8×103/mm3 (normal value 4.5–11.0×109/L), total
bilirubin of 3.2 mg/dL (normal value 0.2–1.2 mg/dL), AST 131 U/L (normal value <40 U/L),
ALT 178 U/L (normal value <40 U/L), ALK phosphate of 339 IU/L (normal value 20–140IU/L).
Urinalysis was negative for infections and a urine drug screen was positive for opiates and
benzodiazepines. Arterial blood gas showed pH of 7.37, PCO2 of 40.7, PO2 of 56,
and a HCO3 of 23.3. Chest X-ray revealed that the patient had multi-lobar
pneumonia () and further workup of her AMS,
which included a brain MRI, revealed a right fronto-parietal and a left cerebellar Infarct
(, ).
Patient was placed on BiPAP due to her hypoxemia, blood cultures were drawn, IV antibiotics
were started, including Vancomycin, Cefepime, and Metronidazole, and patient transferred to
the critical care unit for further management. During the course of her hospitalization,
patient developed severe acute hypoxic respiratory failure requiring intubated. As part of
the work up for the multiple-lobar brain infarct, a trans-esophageal echocardiogram (TEE)
was done within 24 h of diagnosis of multi-lobar infarct to evaluate for cardiogenic source
of ischemic cerebrovascular accident. The TEE revealed a prominent Eustachian valve with
multiple vegetations, diffuse hypokinesia, and a left ventricle ejection fraction of 30%
with a patent foramen ovale as seen in .
Blood cultures revealed Staphylococcus capitis, which was
thought to be a contaminant, but a bronchoalveolar lavage (BAL) culture revealed Methicillin Resistant Staphylococcus Aureus. Patient continued to
improve on medications and was successfully extubated a few days later and mental status
gradually improved, although patient was noted to have some residual deficits that included
a left facial droop and left upper extremity weakness. One week after initiation of
antibiotics, repeat transthoracic echocardiogram showed an ejection fraction of 45–50% and a
normal Eustachian valve. Additionally, repeat blood cultures 4 days after initiation of
antibiotic therapy were negative. Patients symptoms continued to improve and were discharged
to a skilled nursing facility and were set to continue IV Vancomycin and Cefepime for a
total duration of 6 weeks from first negative blood culture. Although a trans-esophageal
echocardiogram could not be repeated due to patient non-compliance repeat transthoracic
echocardiogram was performed at a follow up exam after completion of antibiotics, which
again showed a prominent Eustachian valve but no vegetation, and an ejection fraction that
improved to 55%.


A 66-year-old male with a past medical history significant for UC, diagnosed in 1999 on mesalamine, history of prostate cancer in remission status post radical prostatectomy for 15 years and warm agglutinin AIHA on daily prednisone, who was sent to the emergency room by his primary care physician for evaluation of low hemoglobin. He also has symptoms of anemia, where he complained of increasing fatigue, weakness and shortness of breath on long distance walk. Otherwise, he denied palpitations, headache, and dizziness. He denied also mucocutaneous bleeding, hemoptysis, bloody vomiting, hematuria, hematochezia, and recent infections. He had the upper endoscopy performed in 2017, which showed chronically inflamed duodenal mucosa with extensive foveolar metaplasia and a small focus of gastric heterotopia. He has no history of colonoscopy for the past 10 years.
Initial vitals on admission were stable and physical examination was only notable for scleral icterus and conjunctival pallor. Digital rectal examination and fecal occult blood test were negative. His complete blood counts were significant for elevated white blood cell of 26.06 × 103/µL (predominantly neutrophilic), low hemoglobin 6.0 g/dL, low hematocrit 19.9%, high mean corpuscular volume (MCV) of 133.1, high red cell distribution width (RDW) of 22.6 and low platelet 69 × 103/µL. Initial blood chemistry showed elevated aspartate aminotransferase/alanine aminotransferase 152/83 U/L, elevated total bilirubin 2.2 mg/dL, direct bilirubin was 0.47 mg/dL. His direct antiglobulin test poly Coombs’ test was positive, IgG was positive, C3 was negative, warm auto antibody was positive, PTT was low 20.4 s, PT/INR and fibrinogen were within normal limit. His reticulocyte count was significantly elevated 33.6 %, Haptoglobin was low < 7.75 mg/dL, LDH was elevated 2,894 U/L. iron profile showed normal iron and iron saturation (129 µg/dL, 70.1 %), ferritin was elevated 523.5 ng/mL, and total iron-binding capacity (TIBC) was low 184 µg/dL. Septic workup was negative.
The patient was diagnosed with ES with coexistence of AIHA and ITP. Patient’s platelets count prior to admission was in the range of 200 - 300 × 103/µL. He was transfused with one unit of packed red blood cells (PRBC) and was given 125 mg IV solumedrol in the emergency room. He was continued with solumedrol 80 mg three times daily. Computed tomography (CT) chest/abdomen/pelvis showed mild splenomegaly, which was unchanged from before, there was no lymphadenopathy. The patient was responded to the treatment, his LDH and reticulocyte counts were trending down, and hemoglobin was improved to 7.7 g/dL. Patient was then given IVIG 1 g/kg for two doses because his hemoglobin was still < 8 g/dL. Ultimately, he was discharged with 60 mg prednisone daily for 2 weeks and follow-up in outpatient clinic. On 6 months’ follow-up, his hemoglobin trended up to 12.4 g/dL and platelets improved to 287 × 103/µL.


A 57-year-old male patient presented to the Department of Oral and Maxillofacial Surgery, of a dental institution with a swelling in the left lower lip region for about 6 months. Swelling was uniform in consistency and painless with no other signs of inflammation. Clinical examination revealed an exophytic, nodular, reddish-purple, well-outlined lesion measuring about 1 cm × 1 cm in diameter, located on the left lower lip []. The patient had no other dentoalveolar symptoms and no sign of local infection.
Surgical excision under local anesthesia with diode laser (980 nm) was done. An elliptic incision was made to fully enucleate the lesion along with the overlying mucosa [Figure and ]. Antibiotic and analgesic therapy was given to the patient for 5 days. Antibacterial chlorhexidine gluconate rinse (0.12%) was prescribed for one week.
The specimen was fixed in 10% formalin solution. The histological examination made with hematoxylin and eosin [] and smooth muscle actin staining [] revealed the diagnosis of angioleiomyoma. The lesion was composed of large vascular channels of varying caliber, surrounded by thick walls of irregularly arranged, spindle-shaped cells []. The spindle-shaped cells showed elongated blunt-ended nuclei and eosinophilic cytoplasm-forming smooth muscle bundles showing interlacing patterns with collagen fibers between dilated and slit-like vascular spaces []. Whorled bundles of muscle fibers were seen fused with the vessel walls. Necrosis, atypical mitosis, and pleomorphism were not observed in the histological examination. The postoperative course of the patient was uneventful with 7 days of follow-up []. The tissue was completely healed and there was no sign of scar. There was no recurrence at 3 months of follow-up.


A 62-year-old woman sought medical help in our department for severe low-back pain of 3 years’ duration with accompanied numbness of the right leg. Initially, the patient had no obvious cause of low-back pain and did not go to the hospital for treatment. Application of an analgesic plaster achieved poor curative effect. Subsequently, the back pain gradually worsened, and numbness of the right leg developed at the same time and progressed over the course of 3 years. She visited our department for treatment on March 9t, 2018. She complained that if she sat for more than 2 hours or walked more than 300 m, she experienced low-back pain accompanied by numbness at the front side of the right thigh and lateral calf, and relief of symptoms when she was lying down or bending forward. However, these symptoms seriously affected her daily life and activities.
The patient was subjected to a straight leg–raising test, and the result showed that the right side was positive at 70° and the left side negative. There was no obvious tenderness at the patient’s waist. Patient evaluation was performed using the VAS and Japanese Orthopaedic Association (JOA) scores before and after treatment. In the results Before treatment, scores of VAS of 6 points and JOA of 10 points were obtained. Based on these findings, she was recommended to undergo lumbar magnetic resonance imaging (MRI) to assist in diagnosis. In MRI results (), lumbar curvature was normal and anterior and posterior margins of multiple vertebral bodies rough and sharp. On the T2-weighted image scan, presence of multiple signals of the intervertebral disk, bulging of the intervertebral disk at L2/3, L3/4, and L4/5, protrusion of the intervertebral disk at L5/S1, dural sac compression at corresponding levels, LFH at L2/3 L3/4, and L4/5, and LSS were observed.
Before the operation, positioning and labeling of L2/3, L3/4, and L4/5 with the positioning grid was performed under CT guidance. Subsequently, the CT scan was used to measure the bilateral thickness of the LF at L2/3, L3/4, and L4/5. The angle and depth of the needle on the left and right sides of the L2/3, L3/4, and L4/5 segments were measured on the CT-scan image and the needle-insertion point marked on the body surface (). For routine surgical disinfection, 2% lidocaine was injected into each of the facet joints. The facet joint was punctured with the needle knife, the direction and depth of the needle adjusted to the edge of the lower facet, and the position reconfirmed by CT scan (). Next, the needle knife was used to cut the attachment point of the LF to about 2–3 mm depth and pressed for 10 minutes, and the patient was instructed to maintain a supine position for 6 hours. After surgery, there was a small amount of hemorrhage, no infection, ecchymoma, headache, dizziness, or numbness, and pain and weakness of the lower limbs and other complications (). Then, the thickness of the bilateral LF at L2/3, L3/4, and L4/5 was measured (). In the straight leg–elevation test performed immediately after the operation, negative results on the VAS (0 points) and JOA (10 points) were obtained at both sides. She was followed up after treatment at 1, 2, 4, 6, and 12 months (). In the results of follow-up evaluation at 12 months, VAS of 0 points and JOA of 29 points were obtained. The patient’s waist and leg pain had completely resolved.


A 9-year-old girl presented with worsening cyanosis and breathlessness for the last 3 years. On examination, she was severely cyanosed and clubbed []. The vitals were normal. The clinical examination was otherwise unremarkable with normal second heart sound. There was a hemangioma on the left ear []. There was no family history of similar skin lesions. The chest X-ray and electrocardiogram (ECG) were within normal limits. Her hemoglobin was 16 gm%, and oximetry showed 78% saturation. The liver and kidney function tests were normal. On echocardiography, structurally normal heart was seen. However, a contrast echo showed opacification of the left atrium after 3–4 beats and was diagnostic of pulmonary arteriovenous fistulae (PAVF). The chest computed tomography (CT) showed no parenchymal abnormality and a CT angiogram showed the presence of portosystemic shunt (extrahepatic portosystemic shunt or Abernethy syndrome). The portal vein was draining into the left renal vein []. The intrahepatic portal radicles were not seen.


An 8-year-old girl evaluated for worsening of breathlessness for the last 1 year. She was a known case of pulmonary hypertension diagnosed from 3 years of age and was receiving sildenafil for the same for the last 1 year. The patient had undergone a thoracotomy with a wrong diagnosis of aortopulmonary window at 3 years of age elsewhere. She had nephrotic syndrome 2 years earlier and a kidney biopsy showed membranous glomerulonephritis. The patient had previously received steroids for nephrotic syndrome. On examination, her vital status was normal, and there was no pedal edema. A loud pulmonic component of second heart sound was heard on auscultation, but there were no other abnormal findings. Similar to the first patient, she also had a hemangioma over the left ear []. There was no family history of similar illness or skin lesions. Her chest X-ray showed mildly dilated pulmonary artery. The ECG was normal. The echocardiogram showed no other congenital heart disease and normal right and left ventricle function. There was mild tricuspid regurgitation with an estimated pulmonary artery pressure of 70 mmHg. The CT angiogram showed the portal vein communicating to the inferior vena cava with no intrahepatic portal radicles (Abernethy syndrome) [].


A 24-year-old female presented to our hospital with a 5-day history of fever, vomiting, decreased appetite and malaise and a 2-day history of acute pain in abdomen and abdominal distension. There was no significant medical history or any previous surgery. On presenting to our hospital, she was febrile with 102° F, pulse rate was 102/min, blood pressure was 112/68 and respiratory rate was 20/min. On general physical examination, she had no pallor, icterus or lymphadenopathy. Abdominal examination revealed soft abdomen with tenderness and guarding present in the upper abdomen. Her blood investigations revealed increased leucocyte count (17.08 × 103/mm3) with neutrophilia, low platelet count (149 × 103/mm3), increased prothrombin index (18.2 test/13.5 normal), normal lipase and amylase levels and insignificant liver function tests (bilirubin [T] – 0.50 mg/dl and [D] – 0.25 mg/dl, serum glutamic oxaloacetic transaminase – 29.6 U/l, serum glutamic pyruvic transaminase – 15.3 U/l, alkaline phosphatase – 136.8 U/l and gamma-glutamyltransferase – 55 U/l). Ultrasound abdomen showed distended gall bladder with wall thickening with normal common bile duct, suggestive of acalculous cholecystitis. No abnormality was detected on chest X-ray.
Blood culture was positive for Salmonella typhi with sensitivity to ampicillin, cefixime, chloramphenicol, cotrimoxazole and ceftriaxone. In view of peritoneal signs not explained by ultrasonography, computed tomography (CT) scan was planned. Contrast-enhanced CT scan revealed a small area of discontinuity in the wall of distended thickened gall bladder with resultant pericholecystic fluid which was contiguously tracking along the porta and in the left subcapsular hepatic region.
A diagnosis of gall bladder perforation was made, and the patient was taken up for diagnostic laparoscopy. It revealed purulent collection and flakes in Morrison's space, gastrohepatic region and along falciform ligament; and gastric adhesions with anterior abdominal wall and left lobe of the liver. On adhesiolysis, gall bladder was found to be perforated anteriorly with multiple ischaemic and necrotic areas posteriorly on the neck of gall bladder []. She underwent laparoscopic cholecystectomy with peritoneal lavage []. No gallstone was found either in gall bladder or peritoneal cavity. Postoperatively, the patient was managed with culture sensitive antibiotics, pain killers and intravenous fluids, followed by oral intake on post-operative day 2. The subhepatic drain was removed on post-operative day 4, and the patient was discharged in satisfactory condition.


A 51-year-old male smoker with no significant medical history or risk factors for immunocompromised state presented with concern for a 12-month progressing right cheek swelling unresponsive to antibiotics. The patient denied dyspnea, dysarthria, dysphagia, dysphonia and xerostomia. Family history was non-contributory and the patient was married for 12+ years with children.
Computed tomography (CT) with contrast () showed a right parotid gland fluid collection identified within the superficial lobe of the parotid gland with enlarged lymph nodes present in various levels of the neck. Fine-needle aspiration (FNA) revealed a cyst of the right parotid gland. A right superficial parotidectomy with nerve monitoring was performed and a well-circumscribed mass was excised. Pathology revealed a BLEC of the right superficial parotid measuring 3.5 × 2.5 × 2.1 cm.
The patient presented 6 months later with progressive bilateral cheek swelling, more significant on the right side. Repeat CT revealed multiple bilateral parotid gland lesions with the largest located within the right parotid tail (). Positron emission tomography (PET) scan was ordered to rule out other diagnostic considerations including primary parotid tumors such as Warthin’s tumor or non-Hodgkin lymphoma, or secondary malignancy. Bilateral hypermetabolic parotid lesions were identified along with diffuse bilateral cervical adenopathy (). Findings suggested bilateral BLEC which raised suspicion for a potential immunosuppressive state. An HIV panel was performed with a positive result. The patient underwent initial local aspiration and drainage to palliate his symptoms of cheek discomfort and treatment for HIV was discussed. Patient has since been placed on HIV medication for management and his bilateral cysts and lymph nodes have reduced in size at follow-up visits with no signs of further recurrence.


The 29-year-old Caucasian woman with mild pain, photophobia, and decreased vision in the right eye presented to the emergency department. The patient had been observed in the outpatient department because of keratitis and treated with a combination of topical corticosteroids and antibiotics for 4 weeks but showed no clinical improvement. This young woman is a soft contact lens daily user and has no systemic diseases. The patient mentioned that there had been diagnostic corneal scraping, but no pathogenic cause had been identified.
On presentation day, her best-corrected visual acuity (BCVA) in the right eye was 0.4 (Snellen chart). Slit-lamp examination revealed conjunctival injection and paracentral, round corneal opacification with very mild dot-like fluorescein staining. Anterior segment optical coherence tomography (AS-OCT) showed a 135-µm-deep and 184-µm-wide corneal defect with a 167-µm-deep infiltration zone (Fig. ). The corneal thickness in the affected area was 600 µm and filamental endothelial sediment was noticeable. Corneal scraping and contact lens case were sent for microbiological investigation. Laboratory investigation revealed complete blood count, blood biochemical parameters, and erythrocyte sedimentation rate within normal ranges. In addition, serological results showed negative test results for rheumatoid factor, anti-cyclic citrullinated protein, and anti-neutrophil cytoplasmic antibody screen. C-reactive protein level was normal.
Topical treatment of hourly doses of 0.5% levofloxacin, 0.01% cyclopentolate twice a day and tobramycin ointment 4 times per day was administered. Additional systemic treatment of cefazolin and fluconazole (4 mg/kg) was prescribed.
Repeated corneal scraping was performed on day 6. The microbiology laboratory did not identify any aerobic/anaerobic bacterial or fungal culture growth on either of the samples. Because of slowly progressing corneal melting and no improvement, on day 14 fluconazole solution was changed to peroral voriconazole 200 mg twice a day.
On day 21 spontaneous corneal perforation occurred. AS-OCT revealed a narrow perforation channel to the collapsed anterior chamber. To resume anterior chamber volume, the eye was secured with a soft contact lens. On day 23 the anterior chamber was at normal volume, but the iris was stuck in the perforation site. Hyperdense paracentral structure at the perforation place was visualized with AS-OCT. Slowly, conjunctival reaction, pain, and discomfort reduced. On day 29 the contact lens was removed and sent for microbiological examination. The laboratory did not identify any aerobic/anaerobic bacterial or fungal culture growth.
On week 5 the slit-lamp examination showed iris connection with corneal tissues and radial iris vessel ingrown in the cornea from the iris plug area. The anterior chamber depth was stable. On week 11 under hyperdense stroma was 556-µm-wide stable iris-cornea contact (Fig. ). The patient had no ocular complaints and BCVA was 0.7. Intraocular pressure was 18 mm Hg (rebound tonometry).
Systemic tablet voriconazole treatment took 6 weeks but systemic cefazolin took only 2 weeks in total. Topical treatment consisted of 23 weeks of 0.5% levofloxacin, 0.01% cyclopentolate twice a day, and tobramycin ointment 4 times per day.
On week 45 the patient had no complaints. Slit-lamp examination revealed a clear, bright, and smooth corneal surface with paracentral sharp edge opacification. Empty blood vessels from the iris surrounded corneal opacification. Due to the iris plug, the pupil was mildly reacting to the light, but it was round and central. The anterior chamber had an irregular angle from 40 to 20° (Fig. ). BCVA was 0.8 with correction of −2.25 Dsph −1.25 Dcyl × 15. As this correction was not compatible with comfort for binocular vision, the patient does not use astigmatism correction. Still, with correction of −2.5 Dsph monocular visual acuity was 0.7. Intraocular pressure was 19 mm Hg. AS-OCT showed a paracentral hyperdense stromal corneal scar that was 3,404-µm-wide at the endothelial side but 1,334-µm-wide at the epithelial side. There was also 556-µm-wide stable iris-cornea contact.


A 51-year-old woman with no history of smoking was admitted to our hospital because of a chest shadow in the right lung detected during a routine annual check-up. Computed tomography (CT) scan suggested a 14 × 10 mm pure ground-glass nodule in the apical segmental (S1) bronchus of the right upper lobe (). Three-dimensional CT demonstrated that the apical (S1) and posterior segmental (S2) bronchus originated directly from the main trachea, and the pulmonary vein of the apical segmental (V1) bronchus was seen running dorsal to the pulmonary artery, instead of in front of it (). The bronchus anatomy was confirmed by tracheoscopy (). A reconstructed three-dimensional CT image demonstrated the variable bronchus and pulmonary vein ().
The patient was asymptomatic according to normal physical examination and blood test analysis. No distant metastases were detected by bone radioisotopic scanning. The patient was scheduled for right apical segmentectomy with mediastinal lymph node sampling for suspected adenocarcinoma.
Intraoperative scanning showed that the apical and posterior segmental bronchus originated directly from the right wall of the trachea, as indicated preoperatively. First, the pulmonary artery was exposed through the front of the pulmonary hilum and separated by careful dissection. The artery of the apical segment (A1) was then ligated and cut, and the apical segmental bronchus (B1) was isolated and cut. The pulmonary vein (V1) was ligated and cut at the rear of the apical segmental bronchus (). The lymph nodes around the TB were resected, and right apical segmentectomy was completed using end staplers. The patient’s postoperative course was uneventful. The pathological diagnosis was minimally invasive adenocarcinoma with negative margins and no evidence of lymph node metastasis. The patient recovered well and was discharged 5 days post-operation. No problems were observed 13 months after surgery ().
The patient provided informed consent for publication of this case report.


A 79-year-old woman presented to our infection division with hyperpyrexia. She had nephrotic syndrome and diabetes and had been treated with traditional Chinese medicine, hormones and insulin for a long time. Two weeks prior (day 0), she was referred to a local hospital for fever with shivering. A chest computed tomography (CT) scan revealed inflammatory lesions in both lungs. She was treated with cefazolin and levofloxacin, which were changed to imipenem combined with moxifloxacin on day 7 after her blood culture results indicated an Escherichia coli infection; however, her symptoms persisted after 1 week of treatment.
The patient was subsequently admitted to our infection division for further treatment. At the time of admission (day 15), her physical examination results were temperature, 38.2°C; blood pressure, 124/56 mmHg; heart rate, 99 beats/minute; and respiratory rate, 18 breaths/minute. The patient was lethargic and dispirited. Her laboratory examination results were white blood cells, 36,300/µL with 92.2% neutrophils; C-reactive protein, 105 µg/mL; procalcitonin, 0.5205 ng/mL; and interleukin, 60.042 ng/mL. During her hospitalization, her temperature continued to increase to 40°C (day 21). Her blood, sputum and cerebral spinal fluid (CSF) specimens were sent to our department for microbiological examinations. The CrAg LFA yielded a negative result for the serum (IMMY Inc., Norman, OK, USA) and a positive result for the CSF. India ink staining and CSF culture results were both negative. Parenteral voriconazole was initiated as empiric treatment pending diagnostic testing (day 21).
After 4 days and 16 hours (day 24) of blood culturing using an automated blood culture system (BACTEC FX,BD Becton, Dickinson and Company), pathogens were detected microscopically from two aerobic blood culture bottles. Microscopically, the pathogens were gram-positive, budding, round conidia, suggesting Cryptococcus spp. India ink staining of the fluid in the positive blood culture bottles revealed the pathogenic capsules. Microscopically, the pathogens appeared as rounded cells with thick halos (). The strains were isolated from Sabouraud dextrose agar after 24 hours of aerobic incubation and appeared as beige mucoid yeast colonies (). Identification using the VITEK 2 compact automated system (BioMerieux, France) yielded a positive result for C. neoformans. With confirmation of disseminated cryptococcosis, the initial negative serum LFA test result was questioned. Per the manufacturer’s instructions, we serially diluted the serum and processed it using the CrAg LFA test kit. Increasing dilutions showed that the 1:5 dilution was negative, 1:10 and 1:20 were weakly positive, and 1:40 was positive.
Antimicrobial susceptibility tests were performed via the microdilution broth method using ATB FUNGUS 3 (BioMerieux). In vitro antimicrobial susceptibility testing showed that the minimal inhibitory concentrations for the C. neoformans were 5-fluorouracil (5-FC) 4 µg/mL, amphotericin B (AMB) 0.5 µg/mL, fluconazole (FLU) 1 µg/mL, voriconazole (VOR) 0.125 µg/mL, and itraconazole (ITR) 0.06 µg/mL. As per previous studies, the epidemiological cut-off values for C. neoformans were 8 µg/mL for 5-FC and FLU, 0.25 µg/mL for ITR and VOR, and 1 µg/mL for AMB.– Thus, the patient’s antibiotics were changed to AMB plus 5-FC on day 26. After 1 week of treatment, her infection was controlled, and she was transferred to a local hospital for rehabilitation.


A 70-year-old man who was positive for a blood stool test visited our hospital. Colonoscopy, computed tomography (CT) and barium enema indicated quadruple concurrent locally advanced cancers. The first tumor, with observed lumen stenosis, was located in the ascending colon; the second tumor was located in the descending colon and the third and fourth tumors were located in the sigmoid colon (). CT revealed marked intestinal wall thickness in the ascending, descending and sigmoid colon (). Preoperative precise simulation using 3D angiography was performed to determine adequate lymph node dissection along the arteries feeding the tumors and appropriate resection to avoid anastomotic leakages.
We planned the appropriate placement of trocars as shown in , because we wanted to create a single mini-laparotomy for specimen retrieval and extracorporeal reconstruction after lymph node dissection and mobilization of the colon.
During the operation, laparoscopic exploration confirmed the presence of known four tumors with no invasion of the serosa. Subsequently, a right hemicolectomy and sigmoid colectomy were performed laparoscopically. The right half of the colon was separated, and a side-to-side anastomosis between the jejunum and transverse colon was performed, followed by the sigmoid colon, and a colorectal anastomosis between the descending colon and rectum was performed. The resected tissue specimens revealed four tumors (). Histological examination showed that the first tumor in the ascending colon, the second tumor in the descending colon and the third tumor in the sigmoid colon had invaded up to the subserosa, whereas the fourth tumor in the sigmoid colon had invaded up to the muscularis propria ().
According to the American Joint Committee on Cancer/Tumor-Node-Metastasis Staging System, the p-stage was IIIa, T3N1M0. The patient was discharged 15 days after surgery. For adjuvant chemotherapy, the patient chose to take an oral fluoropyrimidine agent for 6 months. Fortunately, there have been no signs of metastasis or recurrence after the operation at 64 months of follow-up.


We present a case of 54-year-old woman with thyromegaly, back pain, dysphagia, and right arm weakness. She immigrated to the United States from Eritrea in 2011. The thyroid enlarged over 3 months to 5 cm with worsening dysphagia. She denied any voice changes or shortness of breath. Fine-needle aspiration biopsy showed Hurthle cell follicular neoplasm, with normal thyroid-stimulating hormone and free T4 results. Positron emission tomography/computed tomography and magnetic resonance imaging scans found a diffusely enlarged 5 × 4–cm hypermetabolic thyroid mass (standardized uptake value, 10.8) and lytic C7-T1 lesions (standardized uptake value, 11.9) with spinal canal narrowing, concerning for metastatic thyroid carcinoma (). Palliative chemoradiation was initially considered for assumed diffuse metastatic disease. However, after consultation with the orthopedics department, it was decided that she would benefit from thyroidectomy with spinal debridement and stabilization, given the mechanical instability of the T1 vertebral body and symptomatic spinal cord impingement.
She underwent total thyroidectomy, partial T1 cervical corpectomy with a trabecular metal cage, and T1-T2 anterior cervical plate/fusion for mechanical anterior cervical support (), followed by a staged posterior transpedicular decompression at T1 for circumferential tumor debulking with a posterior C5-T3 stabilization procedure. Intraoperatively, frozen samples of the erosive vertebral body lesions showed inflammation, purulent discharge, and multinucleated giant cells without evidence of neoplasm. On permanent section, the thyroid mass showed Hurthle cell adenoma without malignancy, vascular invasion, or cellular atypia (). Anaerobic, aerobic, acid-fast bacterial, and fungal cultures were taken and tested positive for mycobacterium TB complex on polymerase chain reaction with positive QuantiFERON-TB Gold (QuantiFERON) results, confirming Pott’s disease. She was prescribed isoniazid, ethambutol, rifampin, vitamin B6, and pyrazinamide. Postoperative follow-up at 6 months showed resolution of pain, dysphagia, and extremity weakness.


A 31-year-old woman with no medical history was hospitalized for attempted suicide by ingestion of cocaine, benzodiazepine, and methadone. Initially, the patient received intensive care for coma and bradypnoea. The initial electrocardiogram (ECG) recorded sinus tachycardia () and the first blood sample analysis revealed a troponin Ic concentration at 0.56 µg/L (normal range < 0.045 ng/mL), creatinine kinase at 182 IU/L (normal range 20–200 IU/L), a white blood cell count of 6000 10.3/mm3 (normal range 4000–10 000 10.3/mm3), and a normal level of C reactive protein. After naloxone administration, the neurological status quickly improved, whereas the patient developed acute pulmonary oedema with no fever or ECG changes. After a rapid response to intravenous diuretic therapy, the patient was transferred to the cardiology department. The measurement of cardiac biomarkers showed troponin Ic at 2.5 µg/L and brain natriuretic peptide (BNP) at 929 ng/L (normal range <100 ng/L).
The first TTE (, Video S1) showed moderate systolic dysfunction with a left ventricular ejection fraction (LVEF) of 40%, hyperkinesia of the apical segments, and akinesia of the basal segments associated with moderate to severe functional mitral regurgitation (). Global longitudinal strain (GLS) was abnormally low at −16.1%. Longitudinal strain was mainly impaired in the basal segment (). A MINOCA related to a basal TTS was suspected in this stable patient with an interTAK diagnostic score of 81. The patient refused cardiac magnetic resonance because of claustrophobia and we choose to perform coronary computed tomography the next day, which revealed healthy coronary arteries and confirmed the diagnosis of MINOCA ().
In the assessment of this probable basal TTS, 24-h urine stress hormones were measured, showing: noradrenaline at 1206 nmol/24 h (normal range 135–620 nmol/24 h), adrenaline at 136 nmol/24 h (normal range 21–109 nmol/24 h), dopamine at 1351 nmol/24 h (normal range 404–2912 nmol/24 h), normetanephrine at 4.1 μmol/24 h (normal value < 3 μmol/24 h), metanephrine at 0.9 μmol/24 h (normal value < 1.4 μmol/24 h). Based on these biological results, a pheochromocytoma was suspected but ruled out after a normal thoracoabdominal computed tomography scan.
At Day 6, TTE revealed an improvement in basal wall contraction and LVEF (50%) and a reduction of mitral regurgitation, which was now moderate (, Video S2). No rhythm disorder was recorded by ECG telemetry and cardiac biomarker levels decreased (BNP at 335 ng/L and troponin at 0.103 µg/L). The patient left the hospital with 20 mg of FUROSEMIDE.
At 30 days, the patient was asymptomatic and TTE (, Video S3) showed complete recovery of LVEF (60%), normalization of wall motion and of GLS (−21.1%) (), and the absence of residual mitral regurgitation. These results confirmed the diagnosis of basal TTS and the FUROSEMIDE was discontinued.


A female patient aged 15 years with her mother reported to outpatient department of pedodontics and preventive dentistry with complaint of broken teeth, pain, and swelling in the upper front region of the jaw since 2 days. Detailed history revealed that patient had fallen on hand pump while playing during school hours. Pain was dull and continuous in nature that subsided on taking medication whereas swelling was diffuse and limited to the upper front region of the lip. On extraoral examination, the soft tissue laceration was present on upper and lower parts of the lip, whereas intraoral examination revealed the presence of laceration on labial mucosa, complicated crown fracture (Ellis class III) with 11 and Ellis class II with 21. The fractured segment of crown with 11 was loosely attached to the palatal gingiva. Intraoral periapical radiograph showed the widening of periodontal ligament with respect to 11 and 21. Clinical and radiographic examination confirmed the Ellis classes III and II fractures with 11 and 21, respectively ().
The patient and her mother were explained about the treatment procedures in detail and written consent was obtained. Single sitting root canal treatment and reattachment of fractured crown fragment with 11 were planned along with antibiotics and analgesics. Prior to this, oral prophylaxis was done. Under aseptic conditions, local anesthesia was administered in the maxillary central incisors region and access opening was carried out with respect to 11, pulp was extirpated and the canal was thoroughly prepared followed by obturation with gutta-percha (). The broken fragment was approximated intraorally for the evaluation of proper positioning of fractured coronal structure, and beveling was done on the fractured fragment and the tooth to increase the retention. The dentin and enamel of tooth as well as fractured segment were acid etched with 37% phosphoric acid (3M ESPE; ScotchbondTM Multipurpose Etchant, USA) for 20 seconds followed by through rinsing for 10 seconds and drying. Bonding agent (3M ESPE; ScotchbondTM) was applied and light cured according to the manufacturer's guidelines. Then fractured segment was accurately reattached to its proper position followed by finishing and polishing (). Occlusion clearance was carefully checked and postoperative instructions were given in detail.
The patient was recalled after 3 months, and she presented with detached tooth fragment with 11. Therefore, comprehensive decision was taken to reattach the fragment with glass fiber-reinforced composite post followed by porcelain fused to metal crown. The fractured tooth segment was stored in 10% formalin till the completion of procedure. Root canal and fractured segment were prepared for adaptation of fiber post-reinforced composite (Tenax Fiber Trans; Coltene Whaledent Pvt. Ltd, USA) followed by etching with 37% phosphoric acid (3M ESPE; ScotchbondTM) to the pulpal wall of the root and fractured crown and then bonded with dual-cure resin (3M ESPE; Rely XTM U200, Germany) under visible light (). The occlusal clearance was evaluated and post instructions were given. The patient was evaluated after 7 days and the tooth 11 was prepared for full coverage crown, i.e., porcelain fused to metal crown. The tooth were temporary and permanent crown were cemented after 2 days using glass ionomer luting cement (3M ESPE; KetacTM Cem Radiopaque, Germany; ). The patient was recalled after 2 weeks for routine checkup; and further postoperative radiograph after 1 month, 6 months, and 12 months showed no pathology with the treated teeth ().


A 16-year-old boy with a history of the right-sided Bell’s Palsy presented with complaints of a severe underbite and excessive drooling on the left side. These symptoms were progressive over several years to the point that the patient could not comfortably close his mouth. The patient had an unremarkable dental history with no previous oral trauma/procedures. On examination, the patient had facial asymmetry at rest, with the normal function of the facial nerve bilaterally. The facial sensation was intact symmetrically in the V1, V2, and V3 distributions to light touch. He had nystagmus on the right lateral gaze and mild weakness of the left masseter. There was no other indication of brainstem or cranial nerve dysfunction. The remainder of the physical examination was normal.
A computed tomography (CT) scan maxillofacial without contrast was performed, demonstrating a 5.2 × 6.6 × 4.3 cm intra-axial mass with compression and displacement of the brainstem []. There was slight dilatation of the third and lateral ventricles representing mild obstructive hydrocephalus and erosion of the base of the skull involving the carotid canal, foramen rotundum, pterygoid canal, foramen ovale, middle cranial fossa floor, and the internal auditory canal []. An underbite occlusion was noted with associated atrophy of the left masticator, mylohyoid, and anterior belly of digastric muscles and underdeveloped left mandible body and ramus [].
CT angiography (CTA) of the head and magnetic resonance imaging (MRI) brain with and without contrast was performed for further evaluation. CTA demonstrated bony remodeling of the left sphenoid and petrous portion of the temporal bones related to the mass with external compression (approximately 50% narrowing) of the cavernous and petrous segment of the left internal carotid artery (ICA) []. MRI brain showed a 5.2 × 7.8 × 5.1 cm heterogeneously extra-axial enhancing mass centered in the left mesial temporal region with involvement of the skull base and left foramen ovale, rotundum and spinosum, encasement and moderate narrowing of the left ICA, mild obstructive hydrocephalus, and marked compression of the brainstem []. In addition, there was associated atrophy of the left masticator muscles with underdevelopment of the left mandible body and ramus [].
The patient underwent a left orbitozygomatic craniotomy for resection. Pathology of the resected tumor was consistent with the World Health Organization Grade I Schwannoma. Postoperative imaging demonstrated near-complete resection with a thin rim of the residual tumor along the lateral dural margin of the left cavernous sinus/lesser sphenoid wing and at the floor of the left middle cranial fossa extending posteriorly to the roof of the remodeled left petrous temporal bone []. He was evaluated by occupational therapy, physical therapy, speech-language pathology, and physical medicine and rehabilitation physicians while inpatient and was discharged to inpatient rehabilitation on a postoperative day 16. The patient experienced the left cranial nerve IV, V, VI, and VII neuropathies, manifesting as abnormal left eye adduction and external rotation, absent left-sided corneal reflex, left-sided facial weakness, left-sided lagophthalmos, and dysarthria/slurred speech. On follow-up, the patient continues to show significant improvement in the left cranial nerve VI palsy and with improvement, though some residual impairment of the left cranial nerve IV, V, and VII function. Despite these deficits, he is functionally independent with normal breathing and swallowing function. Imaging at 12-month follow-up showed the stable residual tumor. He is currently enrolled in a community college in good standing.


A 59-year-old female patient who had undergone surgical resection of Todani type IV CC with AUPBD 10 years ago at outside hospital () was transferred for surgical treatment for intrahepatic cholangiocarcinoma. The first operation was resection of the extrahepatic extrapancreatic CC, so the intrahepatic and intrapancreatic portions of the CC were not resected ().
Two years after the operation, a follow-up computed tomography (CT) scan revealed development of intrahepatic stones and formation of liver abscesses. The intrahepatic stones were removed through percutaneous transhepatic cholangioscopy (PTCS) (). Six years later after the operation, intrahepatic duct stones developed again, and the intrahepatic remnant portion of the type IV CC was further dilated, so stone removal by means of PTCS was repeatedly performed (). Thereafter, the patient did not undergo regular follow-up with imaging studies.
Ten years later after the operation, a follow-up CT scan revealed development of intrahepatic stones and formation of a new mass (). We performed PTCS to remove the intrahepatic stones again and performed direct biopsy of the mass developed within the intrahepatic CC portion (). The tissue biopsy finding was a poorly differentiated adenocarcinoma. Magnetic resonance cholangiopancreatography at one-month intervals showed slow progression of the intrahepatic mass, and 18F-fludeoxyglucose positron emission tomography showed a hypermetabolic intrahepatic cholangiocarcinoma involving the left hepatic duct and hepatic hilum, with metastatic lymph nodes in the periportal and common hepatic artery areas (). The right liver appeared to be rather shrunken, with noticeable hypertrophy of the left liver, but CT volumetry showed that the proportion of the future remnant right liver volume was 56% of the whole liver volume.
Because the liver mass appeared to be resectable, we planned to perform left hepatectomy with redo hepaticojejunostomy. First, the hepatic hilum was meticulously dissected, and the hepaticojejunostomy was isolated and transected (). We then dissected the enlarged lymph nodes around the common hepatic artery and celiac axis. Thereafter, the right hepatic artery and main portal vein were isolated. After clamping of the left hepatic artery and left portal vein, we assessed the extent of liver resection by means of the discoloration of the liver surface. We performed hepatic parenchymal transection under left hemi-hepatic inflow block without Pringle maneuver. The surface of the dilated intrahepatic CC portion was exposed (). At this step, we opened the right edges of the exposed CC portion with electrocautery to see the luminal structures (). After thorough inspection of the intraluminal mass within the CC portion, we identified the proper transection plane of the right-sided CC and then transected the CC portion along the resection design. The edges of the right-side CC wall were sampled for frozen-section biopsy. Four samples of the resection margins of the CC wall were tumor-negative. Hepatic transection was continued to remove the left liver with cutting of the left hepatic artery and portal vein (). The Spigelian lobe was preserved because its bile duct was not involved ().
The Roux-en-Y jejunal limb, which was made at the first operation, was reused for redo hepaticojejunostomy. Because the wall of the right-side residual CC wall was markedly thickened, we applied multiple fixation sutures along its circumferential edges to facilitate anastomosis and to prevent bleeding at the anastomotic site (). We performed a large hepaticojejunostomy with multiple running sutures of the posterior wall anastomosis and multiple interrupted sutures of the anterior wall anastomosis (). The intrapancreatic remnant CC portion was not resected, because it was not possible to completely remove the remnant intrahepatic CC portion within the right liver.
The pathology report revealed that the mass was a 4.5-cm-sized papillary cholangiocarcinoma with moderate differentiation, which was located at the left hepatic duct (). The tumor extended beyond the bile duct with 7-mm-thick wall invasion from the surface epithelia, but the hepatic parenchyma was not involved. Lymphovascular invasion was present, but perineural invasion was not identified. There was lymph node metastasis in 4 of 15 lymph nodes. The patient recovered uneventfully from this second operation () and is currently undergoing adjuvant chemotherapy for three months.
Considering the clinical sequences of this patient, the reasonable extent of resection seemed to be complete resection of the extrahepatic CC including the intrapancreatic portion combined with proximal partial excision of the intrahepatic CC portion to widen the hepaticojejunostomy opening as much as possible ().