We observed a 22-year-old male patient with marked darkening of the skin, especially on the palms and areolae, jaundice on the skin and sclera, astheno-adynamia, hypotension (80/50 mm Hg), pain in the right hypochondrium. The physical examination revealed asthenic constitution, height 180 cm, body weight 55 kg, reduced facial and body hairs, reduced subcutaneous fat tissue, jaundice on the skin and visible linings, heart rate 105 bpm, enlarged liver, brownish spots on the gums and on the hands. The patient reported that several years ago he was treated with local antimycotic agents for alopecia areata and subsequently received local antimycotics for perioral rash and oral lesions.
The clinical-laboratory investigations revealed increased serum levels of total and indirect bilirubin, AST, ALT, GGT and LDH, low serum sodium and chlorides, but normal potassium, a mild increase in FT4 with increased TSH, very high morning ACTH levels with serum morning cortisol below the normal ranges, and normal testosterone. Whole blood count, biochemical studies, coagulation, glucose, iron, copper, iron-binding capacity, urine sediment, were normal at baseline and during the follow-up. The patient was HBsAg, anti-HBc IgM, anti-HCV and anti-HAV IgM negative.
The immunological investigations showed very low serum IgA levels (0.16 g/l) with normal IgG and IgM, negative ANA, ANCA, AMA, LKM-1 antibodies, antiGAD-60, anti-IA-2, anti-thyroglobulin antibodies, a mild increase in anti-TPO antibodies titer, a marked increase in IgG anti-tissue transglutaminase antibodies – 200 U/ml (), with no changes in cellular immunity (Th, Ts, B and NK cells with normal CD4/CD8 ratio), negative T-SPOT-TB test; HLA type – A*01; B*08; DRB1*03; DQB1*02; karyotype – 46, XY.
The thyroid ultrasound examination revealed normal homogenic glandular parenchyma, thyroid volume 7.8 ml. The abdominal ultrasound failed to visualize the adrenal glands. The liver biopsy showed no inflammation, no fibrosis.
The computed tomography (CT) of the brain revealed normal position and symmetrical narrowing of the ventricular system, narrowing of the subarachnoid spaces, normal hypothalamus and pituitary gland.
The patient was diagnosed with partial serum IgA deficiency together with primary hypocorticism (Addison's disease) based on typical skin darkening, astheno-adynamia, hypotension, very high ACTH levels with low morning serum cortisol, no CT changes in the hypothalamus and pituitary gland. He was treated with saline and glucose infusions, Mannitol, Famotidine, intravenous glucocorticosteroids (methylprednisolone 60-80 mg per day), Fludrocortisone 0.1 mg. The patient was discharged with improvement and remained on the following treatment: Dehydrocortisone 5 mg 1 + 1/4 tablet, Fludrocortisone 0.1 mg 1/2 tablet every morning (1/4 tablet in winter months).


A 14-year-old male presented with complaints of defective vision bilaterally for 2 years duration. His visual acuity was 20/80 in the right eye improving to 20/40 with − 5.0 DS/−1.50 D Cyl at 180° and 20/40 in the left eye improving to 20/20 with − 0.50 DS. There was no history of any systemic illness or medication. Diffuse slit lamp examination revealed mild papillary reaction of the palpebral conjunctiva bilaterally. Examination of the cornea in both eyes indicated multiple subepithelial lines in a pseudovortex pattern extending from the inferior paracentral cornea with some of the lines crossing the pupillary axis. The lines were coarser in the right eye. In addition, isolated discrete white crumb-shaped opacities of varying sizes were present in the anterior stroma of both the corneas. The intervening corneal stroma was clear and the peripheral cornea was not involved [Figure and ]. Optical section illumination demonstrated subepithelial location of the linear opacities and a slightly deeper anterior stromal location of the crumb-shaped opacities []. The right eye had mild corneal steeping in the inferior paracentral region, and the corneal curvature in the left eye appeared clinically normal. The corneal endothelium appeared normal. The rest of the anterior and posterior segment findings were normal.
In vivo laser confocal microscopy (Heidelberg Retina Tomograph 2 Rostock Cornea Module) revealed the subepithelial lesions as linear hyper-reflective “vertebra”- shaped silhouetted structures at a depth of 50–80 microns. The crumb-shaped opacities were denser, discrete, present in the region from 60 to 140 microns and more prominent in the right cornea []. Spectral domain anterior segment optical coherence tomography (Spectralis) confirmed the depth of the crumb opacities to 140 microns [].
Corneal topography (Pentacam; Oculus Optikgerδte GmbH, Wetzlar, Germany) of the right eye showed steepening in the vertical meridian which was more exaggerated in the inferior paracentral region. The maximum anterior curvature was 57.8 D around the 3 mm zone inferiorly. Optical pachymetry indicated a central corneal thickness of 450 microns. There was a significant elevation of the posterior float of the cornea. The Belin/Ambrósio Enhanced Ectasia display indicated keratoconus. Corneal topography of the left eye showed the maximum anterior curvature of 45.2 D around the 3 mm zone inferiorly and the central corneal thickness of 522 microns, which were unremarkable.
The patient was the only child born out of a nonconsanguineous marriage. Slit lamp biomicroscopic examination of his mother and paternal grandfather did not reveal any corneal abnormality.
Genetic analysis was performed using 5 ml of the peripheral blood after obtaining additional informed consent. The study protocols adhered to the tenets of the Declaration of Helsinki and was approved by the Institutional Review Board. Controls were selected from healthy South Indian volunteers without any family history of disease. The coding sequence and splice site junctions of TGFBI (BIGH3) gene were amplified from the genomic DNA of the patient and controls, and the mutation was identified by direct DNA sequencing of the samples.
We observed one reported missense mutation p.Arg555Trp and three previously reported polymorphisms (p.Lue269Phe, p.Val327Val, and p.Phe540Phe). R555W mutation has been reported in various ethnic populations, and this mutation indicates the hot spot specificity and pathogenicity for GCD.


A 41-year-old non-smoker, non-Caucasian man was referred to our respiratory clinic with 1 year history of cough, significant weight loss, appetite loss, and intermittent fever. Apart from well-controlled hypertension, he had no other medical illness. There was no history of contact with tuberculosis patient, and he denied any high-risk behaviors. He had seen three physicians from as many hospitals and on each occasion following pleural , and chest tube insertion was told to have pleural effusion secondary to non-tuberculous infection. CT Thorax was not carried out during his previous hospitalizations. On examination, he was a thin man with a body mass index of 22. There was no finger clubbing, dilated vein, or palpable lymphadenopathies. Respiratory examination revealed features suggestive of right-sided pleural effusion with reduced chest expansion, reduced vocal resonance, reduced vocal fremitus, and stony dullness on percussion over the right mid zone and lower zone. Plain posteroanterior (PA) chest radiograph revealed a huge mediastinal mass obscuring the right heart border with pleural effusion. Contrast enhanced CT Thorax showed a well-defined homogenous anterior right mediastinal mass with adjacent lung collapse as well as pleural effusion with passive lung collapse posteriorly. Note that the cystic nature of the mass could have been mistaken for a loculated pleural effusion (Fig. ). There was mild leukocytosis (total white cell counts [TWCCs] 11.7 × 103/μL), with normochromic normocytic anemia (hemoglobin [Hb] 10.9, mean corpuscular volume [MCV] 80), and his renal and liver profiles were normal. His inflammatory markers were raised, C-reactive protein (CRP) 32.7 and erythrocyte sedimentation rate (ESR) 97. Based on the findings just discussed, he was then referred to the cardiothoracic team for further surgical intervention. At thoracotomy, a cystic-like mass consisted of cloudy fluid measuring 10 cm × 8 cm × 3 cm with generalized adhesion to surrounding lung tissue was found (Fig. ). Histopathology revealed benign cystic teratoma with no evidence of malignancy. Pleural fluid found intraoperatively in the pleural space grew mixed growth of three types of gram positive cocci, most likely because of previous repeated thoracenteses and previous parapneumonic effusion. Postoperative serum alpha-fetoprotein (α-FP) and beta-human chorionic gonadotrophin (β-hCG) were normal, but the recovery phase was complicated by recurrent post-thoracotomy wound infection, which resulted in wound dehiscence. He was initially treated with prolonged intravenous second generation cephalosporin and surgical drainage, but the lesion showed no improvement and, in fact, later had become worse. The infected wound tissue was subsequently sent for direct smear, which showed presence of acid-fast bacilli > 50/L (heavily positive), and the tissue culture grew M. abscessus.


A 57 year-old Chinese female with no other past medical history of significance, presented with recurrent epistaxis and was found to have thrombocytopaenia. Subsequent investigations showed infiltration of the bone marrow with clonal plasma cells with plasmablastic morphology. Although she had mild renal impairment, there was no hypercalcaemia or anaemia. Her skeletal survey was normal; however, there were fluorodeoxyglucose (FDG)-avid bone lesions on PET-CT. She was diagnosed with ISS stage II, IgG kappa multiple myeloma (MM) with normal cytogenetics. Flourescent in situ hybridization (FISH) was not done. Serum M protein was 61.8 g/L at diagnosis.
She underwent 4 cycles of bortezomib, cyclophosphamide and dexamethasone (VCD) and achieved very good partial remission (VGPR). Thereafter she underwent ASCT with a reduced dose of melphalan (140 mg/m2) due to renal impairment. She was subsequently treated with monthly bortezomib maintenance but developed peripheral neuropathy during the fifth month of maintenance after SCT. Her main symptoms were paraesthesia and numbness in the palms and soles which worsened despite symptomatic treatment and discontinuation of bortezomib.
She further developed right upper and lower limb weakness. Her neurologic examination revealed decreased proprioception in a glove and stocking distribution, areflexia, bilateral foot drop and power of 3/5 in the right arm and leg. An MRI brain showed lytic bone lesions with dural-based masses within both occipital lobes. Lesions suspicious of myelomatous involvement were also detected in the pituitary, hypoglossal canal, cavernous sinus and sella turcica. Her MRI spine did not show any myelomatous involvement. Cerebrospinal fluid (CSF) cytology revealed 88% plasma cells which were confirmed by flow cytometry. Her CSF plasma cell morphology and flow cytometric immunophenotyping are shown in respectively. She underwent concurrent cranial irradiation with intra-thecal (IT)-methotrexate/cytarabine and thalidomide-dexamethasone (Thal-Dex) for control of CNS disease. She remained stable for 6 months after relapse, while on lenalidomide and dexamethasone (Len-Dex). She developed progressive disease thereafter, with a rising serum Free Light Chain ratio (sFLC) and multiple FDG-avid PET lesions. She is currently receiving salvage therapy.


A 47 year-old man was admitted to our emergency department after a motorcycle accident with his right hip in a fixed position of 90° flexion, 90° abduction and externally rotation. The patient had a history of hitting an electric pole with his right thigh with abduction force during collision. The patient complained of severe pain during movement of his right hip joint. His vital signs were stable. There were no neurovascular deficits in the injured limb. An anteroposterior radiograph revealed an inferior, obturator-type dislocation of the right hip joint (). Computed tomography (CT) scan was performed before any manipulation to evaluate any associated injury. There was no additional damage to the femoral head, acetabulum or trochanteric area (). After appropriate consultations, manipulation under anesthesia was taken.
General anesthesia was administered with patient in supine position. The dislocation was reduced as per the recommended techniques. We applied continuous traction in line with femur with gentle flexion and lateral push was given on the inner thigh with the pelvis stabilized by an assistant. However, this was unsuccessful. The hip was then internal rotated and adducted. Forceful resistance was abruptly reduced with audible "clunk" and we checked stability and range of motion of the hip. Reduction of the hip joint was confirmed under fluoroscopy and skin traction was applied for maintenance. Post-reduction CT was taken in order to confirm the congruent reduction. Although the post-reduction X-ray had shown minimal contour change of the greater trochanter (), post-reduction CT scan revealed the fracture of the greater trochanter (). The authors considered the possibility of further displacement of the greater trochanter, because the fracture gap was more than 2 mm on CT image. The fracture of the trochanter was then fixed through the incision under fluoroscopy control with cannulated screws and washers (). The patient was discharged with advice for strict bed rest for three weeks, followed by crutch walking for next three weeks. At the final follow-up after two years from trauma, the patient had a pain-free hip joint with active full range of motion ().


A 62-year-old male presented to us with headache and instability beginning a week before, with no other associated symptoms. An emergency CT scan showed a cerebellar mass. There were no medical antecedents of interest, except for previous surgery for an olfactory groove meningioma 11 years earlier without any complications. On admission, blood count showed leucocytosis (12.1 × 10E3/uL) and neutrophylia (91.5%), a globular sedimentation rate of 60 mm/h, and C reactive protein of 1.80 mg/dl. A detailed laboratory investigation revealed that this patient was immunocompetent. A chest x-ray and a thoracic CT scan showed a cavitated lesion at the upper lobule of the right lung [].
Brain magnetic resonance imaging (MRI) revealed a well-demarcated mass of 3.6 cm in diameter located at the right cerebellar hemisphere surrounded by vasogenic edema, hypointense in T1, and hyperintense in T2 []. The images were of cystic appearance, bilobulated, with a fine wall that enhanced after injection of gadolinium []. A body extension study ruled out any other metastatic lesion.
A suboccipital craniotomy was performed, and a cerebellar mass with a necrotic or abscessified content was removed. The histological study disclosed a cerebellar abscess and capsule.
A sample of cerebellar tissue obtained at the operating room was sent to the microbiology laboratory and cultured in standard medium for bacteria and fungi under aerobic and anaerobic conditions at 35°C for 10 days. At 48 hours on the aerobic plaques, small grayish colonies, later identified as ramified Gram-positive cocobacilli positive for urease and catalase were evident. Sensibility tests were performed by E-test®(BD Biodisk, Sweden), with the following minimum inhibitory concentrations (MIC in mg/ml): trimethoprim–sulfamethoxazole 0.064, levofloxacin 0.008, erythromycin 0.125, vancomycin 0.032, imipenem 0.25, linezolid 0.016, daptomicin 256.
Linezolid (600 mg/12 h) was given to the patient with clinical and neurological improvement in the following weeks.
The chest x-rays performed after surgery showed an increase of size of the lesion. Sixteen days after surgery, the patient showed a sudden decrease of consciousness, with a GCS drop from 15 to 10.
An emergency CT scan showed cerebellar swelling and acute hydrocephalus. The patient was immediately operated upon, placing a right external ventricular drainage.
This second time Nocardia was again isolated by the same techniques in cephalorraquid fluid alter 17 days of culture. MIC of tested antibiotics was similar except for imipenem (32 mg/ml). Isolates were sent to a reference laboratory and later identified as N. farcinica by 16S rRNA sequencing.
Antibiotics were changed to vancomycin, imipenem, and ciprofloxacin. Intraventricular vancomycin (1,000 mg/12 h) was also administered.
Chest x-rays showed improvement of the lung lesion, and a transient clinical improvement, but headache recurred after attempts to close the drainage. A control MRI showed a new cystic collection with ring enhancement after injection of gadolinium at the surgical cavity with diffusion restriction at the lower part of cyst that suggested abscess. The patient was re-operated, and an abscess was again found and removed. Again, N. farcinica was identified at the purulent collection. Antibiotic therapy was changed to linezolid and ciprofloxacin following culture results. The patient improved favorably and has been asymptomatic until the last control, 14 months later. The control MRIs showed changes due to surgery with no relapse signs [].


A 56-year-old female presented with an asymptomatic nodular lesion on the trunk. The mass was present since 3 years and had gradually increased in size. Clinical examination revealed a single nodule of size 3 cm × 4 cm on the lateral part of abdomen. The nodule showed hyperpigmented beaded border and pinkish center. It was dome-shaped in appearance and had surface telangiectasia []. Minimal scaling and crusting was seen on the surface of the nodule. There was no significant lymphadenopathy or hepatosplenomegaly.
A biopsy from the nodule showed two different histological patterns, nodular at one half and anastomosing pattern at the other half []. Both the tumor patterns were connected with the surface epidermis and also with one another. Nodular masses were made up of basaloid cells with peripheral palisade, and cleft was noted between the tumor islands and the stroma []. Large aggregates of melanin pigment were seen in the nodular zone. These features were suggestive of nodular BCC. On the other half, cords and columns of basaloid cells were seen forming reticulated network. At places peripheral palisading, melanin pigment, and follicular germ-like structures protruding were seen []. These features were suggestive of fibroepithelioma of Pinkus. Thus the histopathological and clinical features were suggestive of nodular BCC in continuity with fibroepithelioma of Pinkus.


A 55-year-old woman presented with erythematous, violaceous papules, plaques, and annular lesions over her upper and lower limbs since 2 years. Lesions initially started as erythematous papules over both lower legs that resolved after taking oral prednisolone. There were recurrent episodes of healing with hyperpigmentation. There was no history of systemic symptoms.
On examination there were multiple erythematous to violaceous papules, plaques, and annular lesions, and purpuric lesions over both upper and lower limbs. Lesions were bilaterally symmetrical. Healed hyperpigmented macules and atrophic scars were present over the lower legs. There were dry necrotic ulcers over elbows and knees [Figures and ].
Routine laboratory investigations were normal except elevated erythrocyte sedimentation rate (40 mm 1st hr). Antinuclear antibody titers, retroviral and hepatotropic viral serologies were negative. The Venereal Disease Treatment Laboratory test was nonreactive. Antistreptolysin O titers were <200 mIU/mL. Chest radiograph, electrocardiogram, and Mantoux test were normal and slit skin smear was negative. Histopathology of violaceous plaque revealed leukocytoclastic vasculitis; dilated, thickened dermal blood vessels with prominent endothelial cells, neutrophilic infiltration within and around the dermal blood vessels, occasional eosinophilis, nuclear dust, and fibrin deposition around blood vessels [Figures and ]. Serum protein electrophoresis showed M-protein band in beta-2 zone []. Serum immunoelectrophoresis was suggestive of IgA-lamda monoclonal gammopathy []. Urinary Bence–Jones proteins were negative. Bone marrow aspiration was normal. Axial skeletal survey showed no lytic lesions or osteoporosis. Serum calcium levels were normal. She was diagnosed as a case of EED associated with IgA monoclonal gammopathy. Treatment was started with dapsone 100 mg/day orally. Healing of lesions was observed after 48 h of treatment [Figures and ]. Lesions recurred after 2 weeks, and she was started on doxycycline 100 mg/day. Response was obtained with healing of old lesions and no new lesions.


A 66-year-old Caucasian man with decompensated heart failure, asthma, chronic obstructive pulmonary disease, post-traumatic stress disorder, and atrial fibrillation on anti-coagulation was initially referred for management of post-cholecystectomy biliary strictures, which were treated with endoscopic retrograde cholangiopancreatography, and placement of biliary stents. Magnetic resonance imaging performed at that time showed an incidental pancreatic head mass. Subsequently, he underwent (EUS), which demonstrated a 14×12 mm, well defined, hypoechoic lesion within the pancreatic head. EUS-guided fine needle aspiration demonstrated a monomorphic population of neoplastic cells diffusely and strongly positive for CD56, synaptophysin, and chromogranin, compatible with a neuroendocrine tumor (). An octreotide scan showed no evidence of metastatic disease. As the patient was asymptomatic, he was followed conservatively for a non-functioning insulinoma. Six months later, however, the patient was triaged for fasting hypoglycemia (blood sugar levels as low as 45 mg/dL) in tandem with neuroglycopenic symptoms. His C-peptide level was noted to be 7.47 (normal range, 0.80 to 3.85) and his insulin level was 32.8 mIU/L (normal range, 1.9 to 23.0) at the time of hypoglycemia, consistent now with a functioning insulinoma, prompting intervention. Medical therapy with diazoxide (50 mg twice daily) resulted in hypotension, and acute kidney injury necessitating discontinuation. Treatment with octreotide (150 mcg subcutaneously three times daily) did not resolve the hypoglycemic episodes. Although the tumor was resectable, the patient was deemed inappropriate for surgery due to his comorbidities. EUS-guided ethanol ablation was therefore considered. The lesion was injected with 1 mL of ethanol (in 4 divided doses) using three-dimensional targeting, resulting in a hyperechoic blush within the margins of the tumor ( [available online at
]). Following the procedure, the patient’s fasting glucose, insulin level, and C-peptide normalized. He had no post-procedural complications or further hypoglycemic attacks throughout 6 weeks of follow-up, confirming complete remission.


A 38-year-old woman presented with a 10-year history of a productive refractory cough with yellow sputum. Exacerbate symptoms along with low-grade fevers, anorexia, and fatigue were present 4 years before presentation. She did not complain of hemoptysis, chest pain, dyspnea, or night sweats. She had a history of depression and parageusia.
Bilateral equal breath sounds with fine rales but no wheeze were found on lung auscultation. Her blood report revealed a mild normocytic anemia with hemoglobin of 9.6 g/dL. Levels of serum electrolytes and tumor makers, tests of hepatic and renal function were all with normal limits. Pseudomonas aeruginosa were cultured from 3 sets of sputum culture. Repeated sputum samples for tuberculosis were negative. Pulmonary function test revealed a mid-range obstructive ventilatory defect with mildly reduced diffusion capacity. Chest computed tomography (CT) showed extensive bronchiectasis, along with multiple sharp bordered nodules, the largest of which was 15 mm in diameter on the left lower lobe. In addition, stenosis of the left lobar bronchus was seen (Fig. ).
Bronchoscopy performed after admission showed several solid lesions covered with grey granulation tissues in the left primary bronchus as well as in the right upper and lower lobe bronchus, accompanied with peripheral mucosal congestion and edema (Fig. ). The Bronchoscope could not enter the left lower bronchus due to stenosis. Bronchoscopic biopsy revealed chronic mucosal inflammation. Scrubbing slides were negative for acid-fast bacilli.
A neglected history of Chinese medicine kept-in-mouth while sleeping since the last 10 years in this patient provided clues for a final diagnosis. The patient used Coptis chinensis, a Chinese herbal medicine with an extraordinarily bitter taste, to alleviate discomfort of parageusia. The bronchoscope entered the left lower bronchus on the second session. A lot of necrotic tissues and purulent secretions adhering to the ostium of left lobe bronchus as well as right lobe bronchus were seen. A scrap of hard material was clamped during biopsy, which was confirmed to be a foreign body eventually (Fig. ). Subsequently, bronchoscopy clamped out the foreign bodies from each segmental bronchus piece by piece except left upper segmental bronchi. Bilateral tracheobronchial foreign bodies caused by recurrent inhalation of Coptis chinensis is the final diagnosis (Fig. ).
After almost all of the aspirated Coptis chinensis were successfully removed and intravenous antibiotics administered, the patient's symptom improved. However, she still had some productive cough during subsequent follow-ups. Follow-up chest CT revealed severe bronchial dilatation in left lower lobe 10 years after bronchial foreign body was removed (Figs. C, D).


A 35-year-old male, diagnosed with obsessive-compulsive disorder (OCD) for the past 5 years was prescribed on fluoxetine up to 80 mg/day. He was receiving this treatment for the past 1 year with partial improvement of symptoms despite adequate compliance. Cognitive behavior therapy was not possible due to transportation inconvenience. Due to the persistence of symptoms, despite a high dose of fluoxetine, augmentation with clomipramine (up to 100 mg/day) was done. After few days of the addition of clomipramine, the patient had reported dark pigmentation over face localized to both malar eminences. Other than hyperpigmentation, there were no other dermatological complaints. The dermatological consultation was sought for this hyperpigmentation; a diagnosis of “melasma” was made.
His routine hemogram, thyroid function test, and serum cortisol levels were within normal limits. Due to poor tolerance to clomipramine (excessive sedation and severe constipation), dose of clomipramine was reduced to 50 mg/day. Due to the persistence of OCD symptoms, the patient was switched from fluoxetine to fluvoxamine (200 mg/day). This resulted in improvement in OCD symptoms over next 3 months; however, the skin pigmentation and severe constipation persisted. Clomipramine dose was reduced to 25 mg/day for next 2 months and persistence of constipation resulted in its stoppage. Melasma resolved after stoppage of clomipramine. Assessment score on Naranjo adverse drug reaction (ADR) probability scale was six, which was suggestive of probable ADR.[] Assessment on the World Health Organization-Uppsala Monitoring Centre (WHO-UMC) system for standardized case causality assessment also suggestive of “probable” ADR with clomipramine.[] The adverse effect was reported to Pharmacovigilance Programme of India with ADR report no. 2016-29974.


An 86-year-old man presented with a 5-month history of a severe dermatitis affecting the entire body. The patient was not in acute distress, but appeared unwell and exhibited chills. There was no lymphadenopathy present. He denied any other systemic symptoms. A symmetrical, diffuse erythema was present with overlying scale covering >90 % of the body surface area. His skin was warm to the touch. There was no mucosal membrane involvement. The rash began in the groin and had since spread to the rest of the body. It was previously treated with several over-the-counter creams with no amelioration of symptoms. Silodosin had been started for prostatic enlargement 2 weeks before the rash began. In addition to silodosin, the patient was taking ramipril, cyclobenzaprine, atorvastatin, rabeprazole, levothyroxine, meloxicam, hydrocortisone valerate, an antihistamine of unknown name, ipratropium, and bacitracin/polymyxin (exact formulation of Polysporin® unknown). The patient was unable to provide the dosage of any of his medications. With the exception of silodosin, the patient did not report having recently started any of his other medications. Past medical, dermatologic, and family history was unremarkable. The patient quit smoking approximately 2 months prior. Figures and are photographs of the patient’s initial presentation.


A 28-year-old male patient sought the emergency facility complaining of fever over the previous 2 weeks accompanied by headache and malaise. Up to this point, he had been treated with an antipyretic. Three days earlier, he began feeling retrosternal pain radiating to the back, which progressed to palpitations, dry cough, nausea, vomiting, progressive dyspnea, orthopnea, and lower limbs edema before he attended the hospital. He denied previous diseases, regular use of medications, alcohol consumption, tobacco smoking, or use of illicit drugs. Retrospectively, we could not obtain any evidence of promiscuous sexual activity or a history of urethral complaint. He worked in a marble factory and was used to heavy-lifting work without physical constraint.
The physical examination revealed an ill-looking patient who was tachypneic, tachycardic, and febrile (38.3°C), with 120/40 mmHg blood pressure and 86% room air oximetry. His skin, mucosae, and conjunctivae examinations were normal. An auscultation of his heart revealed the presence of an intermittent third heart sound, plus an ejection murmur radiating to the carotids, and a diastolic murmur—the latter two at the base of the heart. Pulmonary auscultation revealed decreased sounds in the right base. The abdominal examination revealed a mildly enlarged liver, and the lower limbs exhibited bilateral edema. Laboratory examinations showed a marked leukocytosis 22,800 leukocytes/mm3 (reference value [RV]: 4.4-11.3 × 103/mm3). There were no other abnormalities in the remaining blood cell count and the biochemical work-up. Urinalysis showed sterile leukocyturia (20,000 leukocytes/mm3). HIV and syphilis serology were negative. The electrocardiogram was normal; the plain thoracic X-ray showed a normal cardiac silhouette and an alveolar space-filling opacity in both inferior pulmonary lobes accompanied by a tiny pleural effusion on the right side (). The transthoracic echocardiogram revealed normal-sized cardiac chambers as well as normal septum and posterior wall thickness, preserved systolic function, and aortic valve thickening with marked diastolic reflux. The patient was hospitalized and an empiric antibiotic regimen comprising vancomycin, gentamicin, and ceftriaxone was started because of the suspected diagnosis of acute infective endocarditis. The outcome was unfavorable with severe hypotension and persistent respiratory failure requiring vasoactive drugs infusion and non-invasive respiratory support, which meant that the transesophageal echocardiography could not be performed. On the second day of hospitalization, the patient presented cardiac arrest and was unresponsive to the advanced cardio life support maneuvers. The autopsy was performed after informed consent by the family.
On the day following the patient's death, one out of three blood cultures was positive for Neisseria gonorrhoeae. Bacterial identification was performed using VITEK-2 compact (version 07.01, BioMérrieux, France) and confirmed by manual biochemical tests. The isolate was resistant to penicillin and ciprofloxacin, but was susceptible to ceftriaxone. Antibiotic susceptibility was determined by disk diffusion according to Clinical and Laboratory Standards Institute guidelines.


A 27-year-old African-American male with a history of schizophrenia, asthma, and cocaine abuse presented to the hospital less than 1 h after intentionally overdosing on 84 mg of iloperidone after smoking crack cocaine to reduce the severity of the post cocaine nadir. There was no independent confirmation of the overdose medication or amount; however, the transient prolongation of the QTc (see below) seems to serve as independent validation of an overdose of some amount. The patient complained of some shortness of breath, but was not tachypneic, was awake and responsive but lethargic, and was tachycardic in 110 s; vitals were otherwise stable and he had an oxygen (O2) saturation of 89% on room air. Electrocardiogram showed a prolonged QTc interval of 527 ms. This dropped to 470 ms (essentially his normal) after 4 days without any arrhythmias. His baseline QTc interval was approximately 440 ms. Since he had a history of developing bronchoconstriction after smoking of cocaine, his respiratory status was a concern. He received intravenous (IV) fluids, oxygen by nasal cannula, nebulizer treatment, and IV steroids. Despite this, his O2 saturation dropped to 78% while on oxygen. He ultimately required intubation and mechanical ventilation and was diagnosed with acute respiratory distress syndrome secondary to complications related to his comorbid conditions and the cocaine use. He eventually recovered without obvious sequelae.


The present case was a 71-year-old male patient who had diabetes mellitus, hypertension, dyslipidemia, and OSA. He had a history of acute myocardial infarction in the mid of the left coronary artery when he was 49 years old. Written informed consent was obtained from the patient to publish patient data.
He developed effort angina at age 63 years and underwent coronary angiography, as three affected branches were found, coronary artery bypass grafting (CABG) was planned. Before CABG, CAVI was 11.8 on the right and 11.5 on the left. His height was 178 cm, body weight was 76 kg, body mass index was 23.9, and systolic/diastolic blood pressure was 135/81 mmHg. He received oral medication with carvedilol 10 mg/day, losartan 50 mg/day, rosuvastatin 7.5 mg/day, aspirin 100 mg/day, and biguanides 500 mg/day; he administered self-injections of insulin – Humalog Mix® 50 (16-18-20 units). CABG was successfully performed. Three years later, at age 66 years, he was suspected of having OSA because of daytime sleepiness and he underwent polysomnography (PSG), which revealed severe OSA with apnea–hypopnea index (AHI) 56.9 events per hour. Continuous positive airway pressure (CPAP) was started at age 66.
During CPAP, the patient had good compliance in wearing a mask from the early stage; during the medical examination, the usage rate was consistently ≥90% (average 7 hours), and residual AHI was maintained at ≤2 events. Five years after the initial CPAP, at age 71, CPAP-PSG was done. shows changes in sleeping parameters between the first and second PSG. After the start of CPAP, apnea disappeared. In addition, the ratio of sleep stage 1 was decreased, while stage 2 and rapid eye movement sleep stage were increased.
An epidemiological study has shown that CAVI increases with age. In the present case, when the patient was 68 years old, CAVI was 10.4 on the right and 10.2 on the left, showing a marked improvement from baseline (age 63; right 11.8, left 11.5) (). CAVI then gradually increased to the baseline level during the subsequent year and a half. Hemoglobin A1c (HbA1c) worsened to 8.9% following an increase in body weight. Owing to an increased dose of biguanides, HbA1c improved, and in response, CAVI also improved. Nevertheless, even after the positive response to treatment with CPAP and biguanides, vascular elasticity increased, as reflected by an increased CAVI, following an increase in body weight due to worsened lifestyle and uncontrolled diabetes. CAVI is little affected by blood pressure level, as shown in .


A 56-year-old male farmer was presented to the emergency department (ED) complaining of right hand discomfort, pain and swelling of 2 days duration. The patient did not report any history of recent trauma. His past medical history was significant for alcoholism and insulin-dependent type 2 diabetes. The right hand demonstrated marked swelling, fluctuation, erythema and associated lymphangitis tracking to the volar aspect of the right forearm. His hand and forearm radiographs were significant for soft tissue edema (). His comprehensive blood work was significant only for an elevated glucose level, however, other signs of infection, including erythrocyte sedimentation rate and C-reactive protein, were absent from laboratory testing.
A localized soft tissue abscess of the right hand was considered and the patient was taken to the operating room for incision and drainage. An abscess involving the middle phalanx of the 5th finger extending to the mid-forearm was incised and drained. Patient was initially administered piperacillin/tazobactam postoperatively. Streptococcus pneumoniae, Klebsiella oxytoca, Pseudomonas aeruginosa and coagulase negative Staphylococcus were identified in the wound cultures that were obtained during the operation. Blood cultures also grew Streptococcus pneumoniae. Despite antibiotic therapy and surgical debridement, the infection progressed to involve the entire fifth digit and amputation of the digit was performed on post-operative day 4.
Despite medical and surgical treatment, the purulent infection progressed and wound cultures confirmed Sporothrix schenckii. The patient was then started on itraconazole after culture results confirmed the organism. During the course of treatment and follow-up, the patient continued to experience right index finger swelling for several weeks. The patient was seen in the outpatient clinic and completed 3 months of therapy. There was no reason to suspect non-compliance in this patient at this time.
Ten months following the course of his treatment, he returned to the ED with complaints of bilateral knee pain, swelling and weight loss. He reported a loss of his insurance and a lack of access to insulin for 4 months. His vital signs were within the normal range and exam findings were notable for 2+ pitting edema and effusions involving bilateral wrists, fingers and knees. An indurated poorly defined 3-cm mass was found in the right popliteal fossa. Significant laboratory values from a complete blood count and comprehensive metabolic panel were glucose 636 mg/dl, C-reactive protein 1.5 mg/dl, white blood cell count 1400 cells/μl and absolute neutrophil count 890 cells/μl. CT scan of the chest, abdomen and pelvis in the ED demonstrated splenomegaly and diskitis involving L4 and L5. The patient was found to have leukopenia but all laboratory tests for the differential causes of polyarthritis were found to be negative including: Antinuclear antibodies (AB), Rheumatoid factor, Anti-citrullinated protein AB, Anti-Ro AB, Anti-La AB, Anti-DNA AB, Serum Protein Electrophoresis, Urine Protein Electrophoresis, Beta-2-microglobulin, Peripheral Smear, Lactate Dehydrogenase, Human Immunodeficiency virus AB, Thyroid Stimulating Hormone and Parvovirus B19 serology. Subsequent bone marrow aspiration indicated only pancytopenia and negative potassium hydroxide preparations and cultures for fungi. CT-guided biopsy of the lumbar spine was performed demonstrating 1+ WBCs but without evidence of organisms, including fungi, through both cultures and staining. Subsequent left hand debridement, right elbow olecranon bursectomy, bilateral open debridement and synovectomy of bilateral knee joints, and removal of left knee mass were performed to evaluate the swelling and pain each location. Cultures from the right elbow and left finger grew Sporothrix schenckii. The patient was started on 3 mg/kg daily dosing of liposomal amphotericin B following the first confirmation of repeat sporotrichosis infection.
In addition to the soft tissue, joint and bone infection with sporotrichosis, pulmonary and ophthalmologic complications also developed during the hospitalization. After 2 weeks into the hospitalization, the patient began to complain about decreased visual acuity of the left eye; ophthalmology was consulted and full examination revealed panuveitis with posterior synechiae. There was concern for endogenous endophthalmitis of the eye, and the patient was taken to the operating room for vitrectomy and posterior synecheolysis of the left eye. Perioperatively, a 12 mm ×9 mm retinal choroidal infiltrate was found, consistent with fungal infection. Cultures, polymerase chain reaction (PCR) and cytology of the vitreous fluid demonstrated not diagnostic which may have been due to the patient having received amphotericin. Additionally, the patient developed epistaxis with hemoptysis, and CT scan demonstrated scattered ground glass opacities throughout the lungs with bilateral pleural effusions, consistent with an infectious process. Thoracocentesis was performed and cultures of the pleural fluid were non-diagnostic. However, the patient continued to improve clinically while receiving liposomal amphotericin.
The patient was discharged following clinical improvement of his conditions, ∼1 month of hospitalization at the time of discharge on oral itraconazole. Despite close follow-up instructions, the patient did not return to clinic and was lost to follow-up.


We report the case of a 25-year-old male, right handed, who suffered from a fracture-dislocation of the left radial head (Mason IV) after a motorcycle incident (). The patient underwent acute surgery for one attempt of open reduction and internal fixation. It resulted impossible for the comminution of the fracture and the surgeons proceeded to resection of the radial head followed by the implant of a press-fit radial head prosthesis (MoPyC, Tornier SAS Etablissement Bioprofile, Grenoble, France), through a postero-lateral access using the Kocher muscular interval. An insufficient intra-operatively stability requested a reinsertion of collateral lateral ligament performed by a Mytec anchor 2.0. The ligament reinsertion and the implant was initially protected and stabilized with an articulated external fixator, maintained for 40 days after surgery (). The intra-operatively evaluation of flex-extension was complete (0–140°). The patient was dismissed without any major complications.
One year after surgery, the patient underwent a clinical, functional and radiological evaluation, under the control of clinicians not involved in the surgery. The patient was also asked to answer the following questionnaires: VAS (visual analogic scale), DASH (disability of the arm, shoulder and hand), MEPS (Mayo elbow performance score) and the Oxford elbow score.
The ROM showed a flexion of the elbow of 140° with a lag extension of 25°, reducible to 20° passively and a complete prono-supination. The elbow was stable on both studied plane. An area of paresthesia was evidenced on the ulnar nerve territory, and the strength showed physiological values. The average results of the scores were the following: VAS 3, DASH 12.5, MEPS 85 and an Oxford Elbow score of 34.
The same patient, at a distance of about 2.5 years from the first traumatic episode, came again to our attention with pain in the left elbow, which appeared after a movement of prono-supination in extension. A rupture of the prosthetic stem was shown on the radiographs (). The patient underwent, as a result, a new surgical procedure. During surgery, after the prosthetic removal, stability tests were performed under anesthesia showing an acceptable stability of the elbow. Based on this evaluation, it was decided not to implant a new radial head prosthesis (). The patient was dismissed without any further complication.
Twenty-seven months after the second surgery, the patient was evaluated investigating all the same parameters listed above. ROM, sensibility and strength showed values similar to those of the previous evaluation (); the paresthesias on the ulnar nerve territory persisted. The varus and valgus stability, evaluated with the elbow in extension, and in 30° and 70° of flexion, appeared similar to the opposite elbow. The new scores showed average results of: VAS 1, DASH 4.2, MEPS 100 e Oxford Elbow Score 46. The scores showed better results than the previous one, and the patient reported a subjective improvement, compared to the time spent with the prosthesis.


A 45-year-old male patient was referred to our pain clinic during admission for abdominal pain control due to chronic pancreatitis. He had been diagnosed with diabetes mellitus type 2, liver cirrhosis and hepatocellular carcinoma due to chronic alcoholism 5 years ago and was being followed up without any viable tumor recurrence after having received transcatheter arterial chemoembolization. His daily medications were 2 mg of glimepiride for diabetes, 10 mg of zolpidem tartrate and 75 mg of trazodone for depression and anxiety, and 10 mg of alfuzosin for benign prostatic hyperplasia. He had made frequent visits to the emergency room complaining of severe abdominal pain, and was admitted to the department of internal medicine for pain control and conservative management.
The patient's chief complaint was continuous epigastric pain and severe postprandial pain of 7/10 on a visual analogue scale (VAS) score of 0 to 10, which was dominant on the left side. The pain itself was well controlled at a VAS of 2/10 with opioid analgesics, but the patient experienced severe constipation and sedation and requested a nerve block in an attempt to reduce his pain medication. He was taking 40 mg of oxycodone four times a day and 8 mg of hydromorphone daily. The patient's most recent abdomino-pelvic CT showed no evidence of tumor recurrence, and atrophic change of the pancreas. Otherwise, there were no anatomical distortions within the patient's abdominal cavity.
After obtaining a written consent form from the patient, we performed an elective left sided unilateral celiac plexus block under fluoroscopy guidance with the patient in the right lateral decubitus position. The patient's pain was clearly dominant on the left side, and because the source of his pain was a benign disease, a unilateral block was chosen in an attempt to reduce any possible complications. A skin wheal was raised with 1% lidocaine at a point 7 cm lateral to the L1 spinous process which corresponded to the inferior edge of the 12th rib, and a 15-cm 22-gauge needle was introduced at a 45 degree angle until it made bony contact with the L1 vertebral body. The needle's angle was adjusted as it was advanced by sliding off the anterolateral side of the vertebral body until it reached the anterior margin of the L1 vertebral body in the lateral view. A 10 ml plastic syringe filled with saline was then attached to the needle hub, and the needle was advanced slowly until loss of resistance was felt and pulsations of the aortic wall could be identified along the shaft of the needle. After the needle was aspirated and found to be negative for blood, a diagnostic block was done with 5 ml of 2% lidocaine mixed in 5 ml of iohexol (Omnipaque™, GE Healthcare Ireland, Carrigtohill, County Cork, Ireland) and adequate spread of dye was confirmed in the anterior-posterior and lateral view radiographs (). The patient was examined for any sensory or motor deficits for 20 minutes, and fluoroscopic evaluation was done to check for any delayed dye spread in unwanted regions such as across the midline of the vertebral bodies in the anterior view and posterior spread or along the psoas muscles in the lateral view. We checked for any numbness or dysesthesia in the T10-L2 dermatomes and weakness in quadriceps function as well as the patient's ability to wiggle his toes. When the patient's abdominal pain had resolved and no other side effects were observed, NCPB was done with 8 ml of 99% anhydrous alcohol (Dehydrated Alcohol Injection, Daihan, Ansan, Gyeonggido, Korea). The patient was followed up for 1 week until discharge from the hospital, and showed no motor or sensory deficits, hypotension or diarrhea. His pain medication was reduced to half of the pre-procedure dose, and the patient's pain was well controlled at a VAS score of 2/10. When the patient came to our out-patient clinic 1 month after discharge, his pain was still well controlled with reduced oral analgesics, but he reported that he had been unable to ejaculate since the procedure. He described his symptom as "a faucet turned off tight", and that he could not ejaculate during climax, which was a symptom that he had not experienced before the NCPB. We referred him to the department of urology and he is being followed-up under the impression of retrograde ejaculation but without any further evaluation. As of now, his ejaculation has not returned during the 3 months period after the procedure.


A previously healthy three-year-old boy presented with a 5-day history of fever (up to 39.5-40℃) and a 1 day history of bilateral conjunctival injection and increasing left neck pain. A physical examination upon admission revealed bilateral conjunctival injection without discharge, fissured red lips, strawberry tongue and tender enlarged left cervical lymph nodes. The largest node, measuring 3×4 cm, was in the left posterior triangle of the neck. Breathing sounds were clear without crackles, and no cardiac murmur was audible. A chest radiograph was unremarkable with a normal appearance of mediastinum and no infiltrates. Initial laboratory results were as follows: erythrocyte sedimentation rate (ESR) 62 mm/h, haemoglobin 12.3 g/dL, white blood cell (WBC) count 16,400/µL (polymorphonuclear leukocytes, 73%; lymphocytes, 19%; monocytes, 5%), platelet count 597×103/µL, C-reactive protein (CRP) 5.8 mg/dL, sodium 139 mmol/L, N-terminal fragment of B-type natriuretic peptide (NT-proBNP) 312 pg/mL. Echocardiography revealed perivascular brightness around both coronary arteries and mild mitral regurgitation. The diagnosis of KD was made at this point, and treatment with intravenous immunoglobulin (IVIG) (2 g/kg/day) and aspirin (80 mg/kg/day) was begun.
Consequently, the fever subsided and conjunctival injection disappeared within 24 hours of IVIG infusion, but the painful left neck swelling persisted and severe torticollis developed. On the 3rd hospital day, the fever reappeared with persistent Kawasaki features. The neck lesion, measuring 4×5 cm, showed diffuse erythematous swelling. It was hard, fixed and hot with extreme tenderness. The follow-up of laboratory results were as follows: ESR 84 mm/h, haemoglobin 10.3 g/L, WBC count 13,510/µL (polymorphonuclear leukocytes, 67%; lymphocytes, 25%; monocytes, 4%), platelets 612×103/µL, CRP 6.9 mg/dL, sodium 134 mmol/L, and NT-proBNP 404 pg/mL. Initial blood, throat, and urine cultures were unremarkable. Normal peripheral blood smear result was noted. A purified protein derivative (PPD) test as well as serologies for Epstein-Barr virus (EBV), and cytomegalovirus (CMV) were also negative. On the fourth hospital day, IVIG was administered again. The fever subsided and his symptoms (injected bulbar conjunctiva, fissured lips and strawberry tongue) slowly resolved until the 6th hospital day. A repeat echocardiogram on the hospital day 6 showed mild dilation of the left main coronary artery (2.5 mm) with trivial mitral regurgitation. Despite defervescence, the patient complained of painful neck swelling and severe torticollis.
On the 6th hospital day, neck computed tomography (CT) showed a low density lesion with an irregular thick wall in the left lateral node, suggesting an abscess, and multiple lymph node enlargements in the left posterior cervical space (). He was thus diagnosed with a combined parapharyngeal abscess and was treated with antibiotics, including ceftriaxone, until the 9th hospital day. However, there was no improvement of the left cervical lymphadenitis.
Because of the lack of improvement of tender cervical adenopathy despite the use of antibiotics, incision and drainage of the presumed parapharyngeal abscess was planned with the patient under general anesthesia on the 9th hospital day. Purulent fluid collection in parapharyngeal space was drained. Shortly thereafter, the cervical adenopathy decreased in size and his torticollis began to resolve. A culture of the abscess revealed the growth of Staphylococcus aureus which is susceptible to clindamycin, erythromycin, and vancomycin. However, ceftriaxone was not included in the antibiotic susceptibilty test. No coronary artery lesion was noted on repeat echocardiogram. On the 14th hospital day, desquamation of the fingertips was noted. A repeat CT scan on hospital day 16 showed decrease of fluid collection (about 0.5 cm) in the left parapharyngeal space compared with the previous CT scan (). The patient was discharged with low dose aspirin and oral antibiotics on hospital day 16.


A previously healthy 26-yr-old male was referred to our hospital because of recently recognized jaundice on May 22, 2008. He complained of anorexia, fatigue, and weight loss of 8 kg over the previous eight months. He lives in the suburbs of Seoul. The medical history was unremarkable except for ingestion of raw freshwater fish. The patient had a blood pressure of 124/81 mmHg, a heart rate of 80 beats/min, a respiration rate of 15 breaths/min, and a body temperature of 36.7℃. The physical examination was unremarkable except for generalized jaundice. The laboratory data revealed white blood cell count 7,100/µL (normal range 4,000-10,000/µL) with 18.3% eosinophils (0-4%), hemoglobin level 12.4 g/dL (13-17 g/dL), platelet count 231,000/µL (130,000-350,000/µL), total bilirubin 11.3 mg/dL (0.22-1.2 mg/dL), direct bilirubin 8.7 mg/dL, aspartate aminotransferase 58 IU/L (< 40 IU/L), alanine aminotransferase 248 IU/L (< 40 IU/L), alkaline phosphatase 563 IU/L (66-220 IU/L), gamma glutamyltransferase 265 IU/L (8-66 IU/L), and amylase 86 U/L (28-100 U/L). Serologic testing of viral markers showed HBs Ag (-), antiHBs Ab (+), antiHAV IgM (-), and HCV Ab (-). The abdominal CT scan showed mild dilatation of the intrahepatic bile ducts (). Because Korea is an endemic area for C. sinensis infection and the patient had a history of ingestion of raw freshwater fish as well as peripheral eosinophilia, we initially suspected biliary obstruction due to C. sinensis infection. Therefore, endoscopic retrograde cholangiopancreatography (ERCP) was planned. On duodenoscopy, the duodenal papilla showed an edematous and bulging configuration with marked hyperemic changes at the orifice (). Cholangiography showed a mild degree of bile duct dilatation with ductal wall irregularities and indentations. However, there was no definite bile duct stricture or intraluminal filling defects, including stones or parasitic adult worms, noted (). We performed endoscopic sphincterotomy for effective bile drainage through the duodenal papilla and sweeping of the bile duct by a balloon catheter. However, no stones or adult worms were identified. We next performed multiple forceps biopsies at the duodenal papilla and inserted a nasobiliary drainage tube up to the second branch of the intrahepatic bile duct for bile collection. Pathology examination of the duodenal papilla showed chronic active inflammation with many eosinophilic cells in the mucosal layer (). In addition, there were parasitic eggs, consistent with C. sinensis found in the drained bile fluid (). Follow up liver function testing five days after the ERCP was dramatically improved: total bilirubin 4.7 mg/dL, aspartate aminotransferase 108 IU/L, alanine aminotransferase 224 IU/L, alkaline phosphatase 336 IU/L, and gamma glutamyltransferase 122 IU/L. The patient was discharged with oral praziquantel (25 mg/kg three times a day) seven days after the ERCP. Two months later after discharge from the hospital, the patient returned for follow up; he was in healthy condition, recovered his body weight and all laboratory studies had returned to the normal range.


A 15-year-old girl visited our outpatient clinic with symptoms of agitation, euphoric mood, flight of ideas, excessive talking, auditory hallucinations, and decreased need for sleep. She was attending an international school in Thailand and had a very good record at school.
Looking at her premorbid features, she fulfilled her responsibility for school life and was sincere in every work. She underwent normal development. In a Thailand international school, she enjoyed school life and had good relationship with friends. Her favorite activities were reading and drawing. She could easily concentrate on something and performed good academic achievement. She had no family history of mood disorder or psychotic disorder.
She had complained of stuffy nose, sore throat, and high fever symptoms 3 months ago, and she had gone to the hospital for a physical examination. At that time, she received a rapid diagnostic test for influenza A virus infection. The doctor suspected an influenza A virus infection and prescribed oseltamivir for her, as 75 mg of oral oseltamivir, twice daily, for five days. Her symptoms improved one day after beginning the medicine.
On her third day taking oseltamivir, the patient became extremely nervous and had difficulty making decisions. However, she completed the 5-day oseltamivir treatment. On that 5th day, she had no need for sleep. She could not sleep at all. She was depressed, experienced loss of appetite, and found it hard to concentrate on conversation and her schoolwork, such as reading books. She experienced a very intense suicidal ideation, reporting that she thought, "How about falling from a balcony?"
The patient visited a psychiatry clinic and received a 2-month prescription for antidepressants. Subsequently, her mood improved slightly and she could sleep. Her suicidal ideation stopped. Therefore, the clinic reduced her antidepressants. A week before her visit to our clinic, her aspect became very different. She felt herself to be the most special person. She felt very confident, with a greatly elated mood. She was more energetic than usual, sleeping less and talking more. She thought that she understood everything. As the week passed, her symptoms worsened, and she began experiencing auditory hallucinations.
The patient visited our psychiatry clinic with the above symptoms, accompanied by her mother. We believed she was in a manic state and prescribed lithium and antipsychotic drugs: 150 mg/day lithium and 100 mg/day quetiapine, to start. We then increased the drug dosage systematically. Her symptoms began to improve within a few days and resolved completely after a month.


A 22-year-old male was transferred to our institution for toxicological evaluation of a presumed black widow envenomation.
The patient’s past medical history was notable for fetal alcohol syndrome and a small congenital ventricular septal defect (VSD). He had no surgical history, except for placement of a chest tube after a traumatic injury five years prior to this presentation. He did not take any daily medications, and he denied using ethanol or illicit drugs but was a tobacco smoker of approximately one pack per day. Despite the history of fetal alcohol syndrome, he was highly functional and employed at a local store.
The patient was sitting outside on a bench at approximately 8 PM when he felt a sharp “poke” on his left thigh and saw a spider on his leg. Shortly thereafter, he developed lower back pain and abdominal pain. Tremors, diaphoresis and bilateral lower extremity paresthesias soon began. He presented to an emergency department (ED) where he was noted to have severe abdominal pain, prompting the clinicians to obtain a computed tomography (CT) of his abdomen and pelvis. His examination was notable for severe diaphoresis, periorbital edema, fasciculations and a target-like lesion over the left thigh. Throughout his 12-hour stay in the ED, he received three liters of normal saline, intravenous morphine, ketorolac, diphenhydramine and ranitidine. Because he was not improving, the decision was made to transfer the patient to our institution for toxicology evaluation.
During the transport, the patient received 100 mcg of intravenous fentanyl. Shortly thereafter, his oxygen saturation was noted to be in the mid 80s, prompting the administration of 2 mg of intravenous naloxone without improvement in the oxygenation. Supplemental oxygen was subsequently administered.
Upon arrival in the medical intensive care unit, the patient was noted to have a blood pressure of 157/94 mmHg and a heart rate of 103 beats per minute. His oxygen saturation was 89% on 4L of oxygen via nasal cannula. His exam was notable for diffuse muscle fasciculations without any muscular rigidity. He was diaphoretic and had distended neck veins with diminished lung sounds. A chest radiograph shortly after arrival revealed diffuse pulmonary edema, and an ECG revealed an incomplete right bundle branch block with ST elevations in the precordial leads (). Laboratory studies were notable for a troponin of 1.37 ng/mL with a CK of 243 IU/L. An arterial blood gas, obtained while breathing 10L/min of oxygen via a simple face mask revealed a pH of 7.33, pCO2 of 42, and a pO2 of 74.6. The patient received 20 mg of intravenous furosemide, with significant diuresis and subsequent improvement in the hypoxia. The patient received one vial of Latrodectus antivenom (“Antivenin [Latrodectus mactans], USP;” manufactured by Merck and Company) intravenously, which resulted in prompt normalization of the vital signs, and resolution of the diaphoresis, fasciculations, and pain.
Approximately two hours after arrival in the intensive care unit, and 15 hours after the envenomation, an echocardiogram revealed a left ventricular ejection fraction of 35–40% with mild to moderate tricuspid regurgitation and a right ventricular systolic pressure of 47 mmHg. No ballooning of the myocardium was noted on the echocardiogram. CT of the lungs revealed no evidence of pulmonary embolus. Repeat cardiac enzymes revealed decreasing troponin values. The patient was started on carvedilol 6.25 mg twice daily. A repeat echocardiogram 48 hours later revealed normalization of the left ventricular function and pressures. The patient was discharged home three days post envenomation.


A 44-year-old male, weighing 79 kg with a height of 176 cm, and presenting with ossification of the posterior longitudinal ligament, was scheduled for multi-level cervical laminectomy using the posterior approach. Aside from pulmonary tuberculosis completely treated 20 years ago, his past history was unremarkable. The patient was newly diagnosed with hypertension and diabetes mellitus after admission, and was placed on anti-hypertensive medication thereafter. A preoperative chest radiograph revealed apical thickening and volume loss in the right upper lobe, and blunting of the right cardiopulmonary angle (). Pulmonary function test did not reveal major obstructive or restrictive pulmonary disease, and echocardiography showed normal sized cardiac chambers and normal valvular function with a left ventricular ejection fraction of 59%.
Upon arrival at the operating theater, standard monitoring devices, including ECG lead II, pulse oximetry and oscillometric non-invasive blood pressure, were applied. Before the induction of anesthesia, glycopyrrolate 0.02 mg was given intravenously. Anesthesia was induced with remifentanil 0.5 µg/kg bolus, followed by 0.1 µg/kg/min infusion, and propofol 1.5 mg/kg. Tracheal intubation with a 7.0 mm (I.D.) wire-reinforced tube (Mallinckrodt Medical, Athlone, Ireland) using a light wand was facilitated with rocuronium 0.6 mg/kg, and the insertion depth was 23 cm at the upper incisors. When both lung fields were auscultated after intubation, only weak, high-pitched sounds were heard with high resistance felt upon manual ventilation. The ventilator, set for a tidal volume (VT) of 650 ml, was able to deliver VT of only 180-220 ml with peak inspiratory pressure (PIP) of 45 cmH2O. Bronchospasm was suspected, so the inspired concentration of sevoflurane was raised to 6 vol%, dexamethasone 10 mg was injected intravenously, and 100% oxygen was supplied. Although the arterial oxygen saturation (SaO2) was maintained at 98-100%, the elevated PIP did not decrease and VT was continuously reduced. An arterial blood gas (ABG) showed pH of 7.309, PaCO2 of 55.8 mmHg and PaO2 of 470.9 mmHg, representing respiratory acidosis. A chest radiograph, taken to rule out bronchial intubation or pneumothorax, showed only the tracheal deviation to the right side and about 2 cm distance between the tip of the ETT and the carina ().
Fiberoptic bronchoscopy was performed, to rule out a thickened mucous plug. Bronchoscopy showed no mucous plugging, however, the beveled end of the wire-reinforced tube was almost completely obstructed by the mucosal wall. The proper position of the ETT could not be verified because of the tracheal deviation. Therefore, the ETT was gradually withdrawn to 21 cm at the upper incisor, and then rotated until the lumen of the ETT became patent under the fiberoptic guidance. The PIP immediately decreased to 22 cmH2O with a VT of approximately 8 ml/kg, a respiratory rate of 10 breaths/min and a positive end-expiratory pressure of 5 cmH2O. After confirming that equal breath sounds were auscultated bilaterally, and SaO2 was maintained at 99-100% with 40% oxygen in air, the patient was turned to the prone position onto the chest rolls. During pinning of the head, the ventilator was again able to deliver a VT of only 180 ml with the PIP increased to 45 cmH2O. Additionally, bilateral breath sounds were again diminished. When the patient was returned to the supine position, the PIP immediately decreased to 22 cmH2O with an adequate VT. As the nearly complete obstruction of the beveled tip of the wire-reinforced tube was considered to be the primary cause, the wire-reinforced tube was replaced with a polyvinyl chloride (PVC) tube with a Murphy-eye side hole (Mallinckrodt Medical, Athlone, Ireland).
Thereafter, the patient was placed prone onto the Wilson frame, and the PIP was stably maintained with adequate ventilation. There was no apparent hypoxemia during the surgical procedures. At the end of the surgery, an ABG showed pH of 7.388, PaCO2 of 35.0 mmHg, and PaO2 of 257.9 mmHg. The patient was extubated and then transferred to the general ward. Chest computerized tomography, taken for postoperative evaluation, revealed a tracheal bronchus supplying the right upper lobe and originating from the right lateral tracheal wall 1.2 cm above the carina (). The patient was discharged on the seventh postoperative day without any pulmonary complications.


The patient was a healthy 29-year-old multigravida. In week 28, the fetus was found to have an anterior neck mass and polyhydramnios was also detected by prenatal ultrasound. During the last four weeks prior to delivery, the neck mass grew substantially, had a broad base, and was 7 cm in diameter. An EXIT procedure was performed at week 38 of gestation by a multidisciplinary team of experts including a pediatric otorhinolaryngologist, neonatologist, obstetrician, two anesthesiologists, and nurses. The patient was placed in a supine position with a left lateral tilt to avoid aortocaval compression. An arterial line for continuous arterial pressure monitoring and two peripheral intravenous lines (16-gauge) were placed before induction. Endotracheal intubation (6.5 cuffed) was done after thiopental sodium (3 mg/kg) and succinylcholine (1.5 mg/kg) administration by a rapid sequence induction. General anesthesia was maintained with sevoflurane 2-3 vol% and nitrous oxide in oxygen (50-50) combined with an additional dose of remifentanil (0.1-0.5 µg/min/kg) and rocuronium (50 mg in total). An end-tidal (ET) concentration of sevoflurane was maintained at 1-1.5 minimum alveolar concentration (MAC) for proper uterine relaxation until the umbilical cord was clamped. Prior to uterine incision, the patient was given two boluses of intravenous nitroglycerine (30 µg), followed by an infusion at 0.5-1 µg/kg/min to ensure uterine relaxation. To maintain a maternal systolic blood pressure of 100-120 mmHg, the following treatments were required: four boluses of intravenous phenylephrine (50 µg), followed by an infusion at 0.05-0.1 µg/kg/min, an infusion of dopamine at 5 µg/kg/min, and rapid infusion of volume expander (500 ml). The skin incision to uterotomy required 22 min. At the time of uterine incision, the obstetrician confirmed that the uterus was fully relaxed and inserted an angiocatheter (20-gauge) into the uterus under the guidance of ultrasound for decompression by slow reduction of the amniotic fluid volume. A uterine incision stapling device was used to reduce intraoperative maternal blood loss. Then, the fetal head and the part of the torso just above the nipple were partially delivered. A sterile pulse oxymetry was applied at the left ear lobe of the fetus after removal of amniotic fluid, but reliable readings could not be obtained. Ultrasound-guided monitoring was done by an obstetrician for the early detection of placental detachment until the fetal airway was secured. The fetus was intubated with a 2.5 endotracheal tube by a pediatric anesthesiologist at the second attempt (). In the first attempt, the stylet of the endotracheal tube was not removed by an assistant due to the slipperiness of the vernix caseosa. The position of the endotracheal tube was confirmed by a sterile end-tidal CO2 monitor. Once the fetal airway was established and the positive-pressure ventilation was satisfactory, the umbilical cord was severed and the neonate was fully delivered from the uterus. Additional resuscitative efforts for the neonate were not required. The time between uterine incision to cord clamp was about 7 min. The 1-min Apgar score of the neonate was 7; the 5-min score was 9. After delivery, the infusions of nitroglycerine, dopamine, and phenylephrine were all discontinued, and the ET concentration of sevoflurane was also lowered to 0.8-1.0 vol%. Then, five units of oxytocin were injected intravenously by bolus, and 20 units/L was infused continuously. There were no bleeding problems after delivery of the neonate. The total operation time was 1 h 55 min. The estimated blood loss was 1,100 ml. The total intake of fluid during the operation was 1,800 ml. Both mother and neonate tolerated the EXIT procedure well. Three days later, the neck mass of the neonate was completely excised by a pediatric otorhinolaryngologist. The excised mass was confirmed to be a 5 × 5 × 5 cm benign teratoma ().


A previously healthy girl of Filipino descent, 2 years and 8 months old, had a 4-week history of rounding of her face, acne on her forehead, an increased appetite, and weight gain of 3.2 kg. Two weeks later, signs of virilization were noted. The maternal grandfather and his brother, both reported to be heavy smokers, died from lung cancer in their 50s. On admission, the patient weighed 16.7 kg (percentile, 95.7) and was 95 cm tall (percentile, 58). Her blood pressure was 142/92 on admission. She had a Cushingoid build with round moon facies and truncal obesity. No abdominal masses were palpable. She had acne on the forehead and increased oiliness of the facial skin. There was no axillary hair and no breast development. The genitalia showed signs of virilization. Laboratory tests revealed 5-dehydroepiandrosterone at 4546 ng/dL (normal, 19–42), testosterone at 84 ng/dL (normal, < 10), and cortisol at 35.1 μg/dL at 8:00 am and 32.3 μg/dL at midnight (normal, < 5). Abdominal computed tomography confirmed the finding of a 4.5 × 4.0 × 4.0 cm right adrenal mass. The mass, involving the right adrenal gland, displaced the kidney inferiorly. The mass was smoothly marginated and appeared fairly vascular with heterogeneous enhancement.
The patient underwent exploratory surgery through a right subcostal incision. The mass was removed intact without violation of its capsule. Postoperatively, her Cushingoid features resolved, and she lost 1.4 kg in 3 months despite adequate oral hydrocortisone therapy. All of her laboratory markers normalized. To date, there has been no evidence of a recurrence clinically, biochemically, or sonographically.


We report here the case of a 53-year-old male patient who had been diagnosed with PKD 9 years previously. The patient was suffering from chronic renal failure and was being treated in the Nephrology Service of Hospital de Base in Sγo Josι do Rio Preto, Brazil. In relation to the intestinal symptoms, the patient was asymptomatic, but had chronic abdominal pain. The patient was progressing to chronic renal failure with for hemodialysis programming.
The patient was admitted to hospital with weight loss and macroscopic hematuria. Frank hematuria began with hemodynamic repercussions and a reduction of all red blood cell parameters, and thus the patient required transfusion. Computed tomography of the abdomen showed multicystic kidneys occupying most of the abdominal space, polycystic liver and probable bleeding of the left kidney that caused a large hematoma [].
The patient was submitted to unilateral nephrectomy. The surgical approach was infracostal and the total surgery time was 2 hours. The right kidney was left in place due to the physical conditions of the patient, but was scheduled for resection during a second procedure. The size of the left giant polycystic kidney was 43 cmΧ19 cmΧ16 cm, with a weight of 4250 g []. An anatomopathological examination confirmed multicystic kidney compatible with renal cystic disease. In the postoperative period, the patient evolved without complications.


A 9-month-old male had undergone the right-sided VP shunt (Chhabra-slit-in-spring silicone shunt) procedure 7 months back for congenital hydrocephalus. He presented to us with complaints that the child protruded a white tube per anus on defecation for past 15 days with clear fluid dripping from it. On examination, the child was afebrile, alert and had no neck rigidity, and the abdomen was soft. On rectal examination, there was a white tube coming from beyond the reach of finger. Total leukocyte count (TLC) was 7200/cumm. An ultrasonography (USG) abdomen was normal. The sigmoidoscopy showed VP shunt protruding into the colon at 18 cm from the anal verge. The child was operated and the shunt was cut at abdominal surface through a small incision. The rest of the distal tube was extracted per rectum. The proximal tube was taken out as external drainage. On antibiotics, the child improved. The cerebrospinal fluid (CSF) culture done after 1 month was sterile and so a revision of shunt was done on the left side. The child was asymptomatic at 3 years follow-up.


A 3-year-old male, who underwent VP shunt 1 year ago for congenital hydrocephalus presented with similar complaints as the above patient []. On examination, the child was stable except that proctoscopy showed VP shunt protruding into the rectum at about 12 cm from anal verge. TLC was 6400/cumm, CSF was sterile and the USG abdomen was normal. The child was operated and managed similarly as the above patient with successful outcome and is on regular follow-up.


A 53-year-old female was blue lighted into our emergency department resuscitation room by ambulance crew as she appeared extremely cyanotic from face to feet. She complained of nausea, vomiting and difficulty in breathing. Her most striking feature was a bluish hue of her face (Fig. ) including her lips and forearms. She looked as if she had fallen onto blue ink and had some residual staining. However, closer examination revealed that this discolouration was present throughout, including her nails (Fig. ). Oxygen therapy had made no difference to her appearance. She appeared confused and was unable to give any coherent history.
She was tachycardic (130/min) and tachypnoeic (30/min), and had a SpO2 of 90% on 15 l oxygen; blood pressure was 100/50 mmHg. Chest examination revealed few crackles in both mid zones, and a possible chemical/apsiration pneumonitis was suspected. She had a history of deliberate self harm. She had no allergies and worked as a beautician by profession.
Arterial blood gas (ABG) analysis on oxygen showed pH 7.41 (7.35–7.45), pCO2 3.44 kPa (4.7–6), pO2 31.6 kPa (11–13 on room air), lactate 11.0 (<2), base excess −7.2 mmol/l (−2–+2) and HCO3 19.4 mmol/l (24–31). The patient continued to receive high-flow oxygen and intravenous access was gained, and bloods tests, including paracetomol and salicylate levels, and a urine toxicology for the usual drugs of abuse were requested. These subsequently turned out to be negative. She was treated with intravenous crystalloids for compensated metabolic acidosis and treated with antibiotics to cover for aspiration pneumonia. A differential diagnosis for high anion gap metabolic acidosis was worked out using the common mnemonic (MUD PILES—Methanol, Uremia, Diabetic ketoacidosis, Paraldehyde, Iron, Lactic acid, Ethanol and ethylene glycol, and Salicylates, respectively) as the anion gap was 21 (3–11). Despite her treatment, there was no sign of improvement. The initial chest x-ray was normal.
A diagnosis of methaemoglobinemia was suspected in our patient as she demonstrated (1) central cyanosis unresponsive to oxygen therapy with no previous history of congenital cardiac disease or causes for pulmonary hypertension; (2) decreased oxygen saturation on the pulse oximeter with a near normal pO2; (3) difference in the arterial blood gas (95%) and the pulse oximeter (90%) oxygen saturation measurements; and (4) she was a beautician with a history of self harm (beauty products when ingested can result in methaemoglobinaemia). An urgently requested methaemoglobin (MetHb) level showed a raised MetHb level (50.3%), which confirmed the diagnosis. She was subsequently treated with a slow intravenous dose of methylene blue (2 mg/kg). Clinical improvement was marked within 20 min. Her tachycardia and tachypnoea began to settle, and she was now able to communicate.
Her repeat ABG, 30 min post treatment, showed pH 7.37, pCO2 4.12, pO2 11.6, lactate 6.4, base excess −6.8, cHCO3 19.2 and MetHb 2.9%. She acknowledged the fact that she had consumed alcohol but refused to answer the deliberate self-harm questions. She was observed in a resuscitation room with regular ABG monitoring through an arterial line and then transferred to the intensive care unit where her recovery was complicated by the development of ARDS. ARDS was confirmed on a CT scan and was thought to be vasogenic or neurogenic in origin rather than aspiration or infective. She gradually improved and was eventually discharged from the hospital about 10 days later. Psychiatric counseling revealed that she had ingested a barbicide, and out-patient follow-up including chest clinic was arranged prior to discharge from the hospital. She was reviewed in the chest clinic for up to 3 months and had made an excellent recovery and did not have any ongoing respiratory symptoms, and hence was discharged from follow-up.


A 50-year-old male patient visited our department on July 14, 2009 with the chief complaint of anterior teeth sloping down and open. As mentioned above, clinical and radiographic examinations were performed ( and , ). He was diagnosed with chronic periodontitis localized in the maxillary anterior teeth area and pathologic tooth migration. The treatment plan was periodontal therapy to eliminate causes, tooth movement of 11, and prosthodontic treatment.
and show clinical photographs of the front side and the occlusal plane after periodontal treatment. As mentioned above, teeth were fixed at the location to where the teeth would shift with wax on the cast after space analysis ( and ), and the clear aligner was fabricated ().
and show clinical photographs of the front side and the occlusal plane after 3 weeks. After space analysis, the teeth were fixed at the location where the teeth will be shifted to with wax again and the clear aligner was fabricated ( and ).
and show photographs of the front side and occlusal plane after 6 weeks. Since the patient was satisfied with the results, use of a retention appliance was planned instead of prosthodontic treatment. After taking radiographs (), the retention appliance was applied ().
As a result, the probing pocket depth (PPD) and gingival recession (GR) decreased about 1 mm, and clinical attachment level (CAL) decreased about 2 mm when compared to the first visit. Also, mobility decreased slightly ().
In the radiographs, vertical and horizontal dimensions were measured (). Radiographically, the size of the infrabony defects decreased 0.8 mm vertically and 0.4 mm horizontally. However, actual bone filling in the lesions could not be determined based on the radiographic view alone.


A 34-year-old male patient visited our department on May 25, 2009, with chief complaints of anterior teeth separating and discomfort in the gums. As described above, clinical and radiographic examinations were performed ( and , ). He was diagnosed with an endo-perio lesion due to loss of tooth vitality and the pathologic tooth migration of 21. The treatment plan was periodontal therapy to eliminate causes, tooth movement of 11 and 21, and prosthodontic treatment.
and are clinical photographs of the front side and the occlusal plane after the periodontal treatment. As in case I, clinical photographs at the front side and occlusal plane views were taken after fabricating clear aligner on the cast and applying the device ( and ). and are clinical photographs of the front side and occlusal plane views after 3 weeks of using the new appliance. A new appliance was applied again after 6 weeks ( and ). Prosthodontic treatment was performed as planned ( and ). After completing prosthodontic treatment, radiographs were taken for radiographic evaluation ().
To measure the results of the treatment, PPD, CAL, GR, and mobility were measured after treatment. GR and CAL decreased about 1 mm compared to the first visit, and mobility decreased slightly ().
In radiographs, the vertical and horizontal dimensions were measured after treatment (). Radiographically, the size of infrabony defects decreased 2.1 mm vertically and 0.7 mm horizontally. However, as in case I, actual bone filling in the lesions could not be determined based on the radiographic view alone.


The third patient was a 54-year-old male who visited our department June 11, 2009, with the chief complaint of anterior teeth sloping. Though clinical examination was performed as mentioned above (), no radiological examination was done due to the absence of the appearance of an infrabony defect (). He was diagnosed with chronic periodontitis localized on the 13-23 area and pathologic tooth migration. The treatment plan consisted of endodontic treatment, followed by periodontal therapy and tooth movement of 11 and 21, and prosthodontic treatment.
After periodontal treatment was performed ( and ), an appliance was inserted ( and ). Clinical photographs of the front side and occlusal plane were taken 6 weeks after appliance insertion ( and ). Though prosthodontic treatment was recommended to the patient, the retention appliance was used. That's because the patient wanted to finish in the current state (). GR and CAL decreased about 1 mm compared to the first visit and there was no change in mobility ().


A 51-yr-old female was referred to our outpatient clinic for the evaluation of generalized edema on November 17, 2009. Two years previously, she had been diagnosed with idiopathic thrombocytopenic purpura (ITP). She had taken no medicine. Except for the ITP, she had no history of systemic disease like diabetes or hypertension. On admission, her blood pressure was 140/80 mmHg with a pulse rate of 76 beats/min. Her lower extremities showed pretibial pitting edema of grade 4. The patient also reported foamy urine for several days.
The chest radiography revealed cardiomegaly (the CT ratio was 0.54) and bilateral pleural effusions. The ECG showed normal sinus rhythm. Both kidneys showed diffusely increased parenchymal echogenicity with partially obliteration of the corticomedullary differentiation and a normal range of size (the right kidney was 10.3 cm by 4.5 cm in size, and the left kidney was 9.5 cm by 4.5 cm in size).
The laboratory findings showed nephrotic syndrome with generalized edema; the 24 hr urine protein was 5.3 g/24 hr, the total cholesterol was 7 mM/L (normal: < 5.2 mM/L) and the serum albumin was 22 g/L (normal: 35-52 g/L). On admission, the fasting blood glucose and hemoglobin A1C (HbA1c) were 5.4 mM/L and 0.05 Hb fraction, respectively. The blood urea nitrogen and serum creatinine were 20.6 mM/L (normal: 2.6-7.3 mM/L) and 0.23 mM/L (normal: 0.05-0.11 mM/L). In addition, the patient had a positive antinuclear antibody titer at 1:1,600 (normal:negative), positive antibodies against double-stranded DNA at 400 IU/mL (normal: < 100 IU/mL) and she was negative for antiphospholipid antibodies. The complement C3 and C4 levels were low at 0.28 g/L (normal: 0.76-1.39 g/L) and 0.073 g/L (normal: 0.12-0.37 g/L), respectively. She was diagnosed with systemic lupus erythematosus by the American College of Rheumatology criteria (). At diagnosis, the SLEDAI score was 14. At the 6th day of admission, a renal biopsy was performed. That showed diffuse global glomerulonephritis with active lesion and IV-G(A) by the ISN/RPS classification ().
Immunosuppressions consisting of high-dose steroid (methylprednisolone 750 mg for 3 days followed by prednisolone 60 mg), cyclophosphamide pulses (500 mg at the 6th and 36th day of admission), as well as plasma exchange were all started. In addition, she had oliguria that was refractory to diuretics, and this led us to start hemodialysis. After four session of hemodialysis, the oliguria was resolved. The clinical course was complicated by thrombocytopenia, which might be due to cyclophosphamide. The peripheral blood smear revealed normocytic normochronic anemia and thrombocytopenia without abnormal cells. There was no clinical evidence of infection or disease flare up.
While maintaining a stable clinical course, a generalized myoclonic seizure occurred at the 47th day of admission. At that time, her body temperature was 36.1℃. The laboratory and neurology studies showed hyperglycemic hyperosmolar syndrome with a blood glucose level of > 44.4 mM/L, the serum osmolarity was 382 mM/kg (normal: 275-300 mM/kg) and the hemoglobin A1c was 0.082 Hb fraction. The serum pH was 7.348 and the bicarbonate was 26.2 mM/L (normal: 21-28 mM/L). The urine dip stick test was negative for ketones. The other electrolyte levels were within the normal limits. Brain MRI and EEG showed brain atrophy without other lesion. The C-reactive protein was normal at 3.9 mg/L (normal: 0.1-4.7 mg/L). The complement C3 and C4 levels were 0.66 g/L (normal: 0.76-1.39 g/L) and 0.237 g/L (normal: 0.12-0.37 g/L), respectively. At shortly before seizure, the SLEDAI score was 12. The patient was treated with insulin and anticonvulsant medication. The seizure stopped after the blood sugar and serum osmolarity declined below the upper normal limit mentioned above. The patient became asymptomatic and she was discharged 10 weeks after admission under maintenance therapy with prednisolone 10 mg, insulin glargine 12 unit and nateglinide 270 mg. On follow-up 1, 2, and 3 month after discharge, the HbA1c was 0.051, 0.049, and 0.06 Hb fraction, respectively. The changes of laboratory findings according to treatment are showed in , . The patient remained asymptomatic under maintenance therapy with deflazacort 12 mg and without insulin or medication for blood sugar control.


A 55-years-old man was referred to our department with a 5-month history of painful masses in the left portion of the neck. The medical history was unremarkable. Two masses were palpated in the right parotid region and submandibular (level IIA) area. Computerized tomography scans revealed a tumor in the right parotid region and another in the submandibular area, both approximately 3 cm in diameter (). The fine needle aspiration cytology along with clinical observation showed that the lesion was similar to a hemangioma. The patient was scheduled for surgery. A modified Blair incision was used to approach the lesion. After exposing the main trunk of the facial nerve and the peripheral branches, purple colored tumors were found at the surface of the parotid gland and submandibular area. The tumors and the surrounding tissues were removed, leaving the facial nerve intact (). Following the operation, the patient did not have severe bleeding or facial palsy. Histologically, the tumor contained many capillaries and a few wide vessels. Glomus cells that were positive for smooth muscle actin using an immunoperoxidase stain were scattered between these cells. S-100 protein, pancytokeratin, desmin, glial fibrillary acidic protein, and HMB-45 were negative in the glomus tumor. Staining for CD34 was positive in the vascular endothelial cells, but negative in the glomus cells. The glomus cells were arranged in organoid and trabecular patterns surrounding vascular lumens. No lymphoid cells or tissues were observed in any of the sections. A diagnosis of glomus tumors was made.
According to the number of glomus cells, vascular structures, and smooth muscle tissue, the tumor located at the submandibular area was classified as an angiomatous type, and designated as glomangioma. The other tumor at the parotid region was classified as a solid type with masses of glomus cells, and was considered as a glomus tumor proper (). The postoperative period was uneventful, and no signs of recurrence have been noted.


The patient was a 51-yr-old woman with swelling of the extremities and bilateral ptosis on March 8, 2010. She complained of exertional dyspnea and weakness. She married at the age of 25. After that time, she developed drooping of her eyes, with the left eye drooping more than the right eye. Her symptoms persisted and they had gradually increased during about the last 10 yr. About 8 yr ago, she underwent a ptosis repair operation at a local clinic, but her symptoms were not improved. She denied a history of loss of consciousness, headache, dysphagia, dysarthria and myalgia. There was no family history of similar symptoms and no history of trauma. There was no previous history of diabetes and hypertension.
On examination, she was determined to be 151 cm in height and 48 kg in weight. The initial vital signs were a blood pressure of 140/80 mmHg, a pulse rate of 68 beats/min and a respiratory rate of 20/min. She was lethargic and had facial puffiness and no goiter. The lung and heart examinations were unremarkable. All the extremities showed pitting. She had marked bilateral ptosis, while the external ocular movements were normal (). The other cranial nerve examinations were unremarkable.
The laboratory findings were a total leucocyte count 4.75 × 103/µL with 61.6% polymorphs, a hemoglobin level of 9.0 g/dL, the random blood glucose was 124 mg/dL, the serum sodium was 130 mM/L, the potassium was 3.7 mM/L, the blood urea nitrogen (BUN) was 13 mg/dL, the creatinine was 0.8 mg/dL, the aspartate transaminase (AST) was 81 IU/L, the alanine transaminase (ALT) was 29 IU/L, the total bilirubin was 0.93 mg/dL, the creatine kinase (CK) was 1,195 IU/L (normal reference: 20-180 IU/L ), the lactate dehydrogenase (LDH) was 517 IU/L (normal reference: 101-218 IU/L), the total cholesterol was 256 mg/dL, the triglyceride was 121 mg/dL, the high density lipoprotein cholesterol (HDL-C) was 40 mg/dL and the low density lipoprotein cholesterol (LDL-C) was 196 mg/dL. The urinary analysis was negative for blood and protein with using a dipstick. As the clinical findings suggested hypothyroidism and myasthenia gravis, a thyroid function test and acetylcholine receptor binding antibody test were done. The serum T3 was 0.195 ng/mL (normal reference: 0.86-2.02 ng/mL), the free T4 was 0.08 ng/dL (normal reference: 0.93-1.705 ng/dL), the thyroid-stimulating hormone (TSH) was 2.08 µIU/mL (normal reference: 0.27-4.2 µIU/mL), the antimicrosomal antibody was 17 IU/mL (normal reference: 0-34 IU/mL), the antithyroglobulin antibody was 20.1 IU/mL (normal reference: 0-114 IU/mL) and the acetylcholine receptor binding antibody was negative.
Secondary hypothyroidism was suspected. We performed a careful history taking and other pituitary hormone evaluations. At the age of 26 the patient delivered her daughter and she had a history of massive postpartum vaginal bleeding. Thereafter she not resumed menses. The basal levels of other hormones were a serum cortisol of 4.03 µg/dL, the adrenocorticotropic hormone (ACTH) was 29.64 pg/mL, the growth hormone (GH) was 0.06 ng/mL, the IGF-1 was 25 ng/mL (normal reference: 71-263 ng/mL), the prolactin was 1.59 ng/mL, the luteinizing hormone (LH) was 1.49 IU/L, the follicle-stimulating hormone (FSH) was 4.91 IU/L, the E2 was 17.29 pg/mL and the testosterone was 0.02 ng/mL. The combined pituitary stimulation test, including the insulin tolerance test, the thyrotropin-releasing hormone (TRH) stimulation test and the gonadotropin-releasing hormone (GnRH) stimulation test showed panhypopituitarism (). Brain magnetic resonance imaging (MRI) showed a finding of an empty sella turcica and there was no evidence of an intracranial mass, hemorrhage and aneurysm ().
Nerve conduction study (NCS) and electromyography (EMG) of the limbs reveled normal results with the exception of incidentally detected carpal tunnel syndrome. Needle EMG of the orbicularis oculi showed no evidence of dysfunction of the neuromuscular junction.
We diagnosed her as having Sheehan's syndrome, bilateral ptosis and subclinical myopathy caused by secondary hypothyroidism. Replacement with prednisolone of 10 mg/day and thyroxine (T4) 100 µg/day was started. The dose of prednisolone was decreased to 5 mg/day after 4 weeks. At 3 months follow-up, she had become euthyroid with normalized muscle enzymes. She recovered from her presenting symptoms and the bilateral ptosis was much improved ().


We present the case of a 68-year-old woman who was diagnosed with ulcerative colitis (UC) in1983. Since then she had frequent flare-ups and was repeatedly treated with courses of steroids. She was receiving 5-amino salicylic acid as a maintenance therapy. She presented to the clinic with a flare-up that coincided with her return from Thailand 22 days earlier. She complained of passing 7-8 loose motions per day and having generalised symptoms of malaise, low grade fever and tachycardia. With these symptoms and a Creactive protein (CRP) of 55, she was hospitalised and placed on intravenous steroids. Stool samples were negative by both microscopy and culture. Despite intravenous steroids, her diarrhoea worsened and became bloody. Sigmoidoscopy showed severe inflammation with cobblestone formation, bleeding, and friability of the mucosa from the rectum to beyond the sigmoid colon, which is not characteristic of UC. Therefore, multiple biopsies were taken from the rectum and sigmoid colon; the histology showed focal mucosal ulceration due to invasive amoebiasis, and E. histolytica parasites were evident (Fig. 2). Steroids were stopped and the patient was started on a course of metronidazole at 750 mg three times daily for 10 days, followed by oral diloxanide furoate (500 mg) three times a day for 10 days. She eventually recovered and was discharged on 5-amino salicylic acid. Since then she has remained in complete remission.


A 76-year-old male was diagnosed with lung cancer without histological confirmation. He was found to have developed a thrombosed aneurysm in the descending thoracic aorta. Thoracic CT scan revealed eccentric aneurysm of descending thoracic aorta with a length of 10 cm, diameter 6.5 cm, filled partially by thrombus ().
The decision was made, considering his general condition and underlying lung cancer, to repair this aneurysm using an endovascular graft. The procedure was performed in the angiography room using regional anesthesia. Femoral artery access was obtained bilaterally via cut downs. The right femoral artery underwent uneventful retrograde cannulation, measurements were performed, and a 40 mm diameter ×12 cm length stent was successfully inserted (SEAL S&G BIOTEC. Seongnam, Korea) distally.- Completion arteriogram revealed a widely patent endovascular graft with patent descending thoracic aorta (). There was no evidence of proximal or distal endo-leakage. The procedure took approximately 3 hours in total, which remained well tolerated and the patient remained normotensive (systolic blood pressure 140-110 mmHg, diastolic blood pressure 110-70 mmHg), and the urine output was maintained constantly throughout. He complained of absent motor function in the extensor and flexor of the hip and both legs, especially on the left side 3 days after the procedure despite his blood pressures were well maintained during and after the procedure (systolic blood pressure 140-100 mmHg, diastolic blood pressure 110-65 mmHg). The motor grade of ankle dorsiflexors was estimated to be approximately 1/5, plantar flexors 1/5 and hallucis longi 2/5. Pinprick and temperature sensation were intact at the T9-T10 level with normal toe proprioception. An emergency MRI was obtained and the findings were consistent with spinal cord ischemia at T6-12, with elongated bright signal lesion on T2WI in mainly gray matter location (). These findings, along with abnormal neurologic examinations, were consistent with the anterior spinal artery syndrome.
The patient was managed with systemic steroid therapy, spinal drainage and pharmacologic support of blood pressure. The patient's mean arterial pressure was maintained using vasopressor at 75-85 mmHg and the lumbar cerebrospinal fluid (CSF) was drained in 10 mL aliquots to maintain CSF 12 mmH2O or less. Glucocorticoid therapy was administered in bolus at 30 mg/kg over 15 minutes, with maintenance infusion of 5.4 mg/kg for 48 hours.- On post-procedure day 8, the CSF catheter was disconnected. His motor strength slowly showed improvement but his ankle flexion strength was 2/5 on the right, and 3/5 on the left. The patient was discharged on post-procedure day 40 with residual neurologic deficit. Two months later, he gained nearly full knee and ankle motor strength at 4/5 in both legs.