A 45-year-old woman presented with pain and swelling in the lower back and buttocks for more than 8 years and had recently developed sciatica and bowel incontinence. There was no history of trauma. On examination, the region over L4-L5 and sacral spine was tender with a diffuse swelling measuring 11 cm × 10 cm. Magnetic resonance imaging scan showed a large heterogeneous presacral soft tissue mass lesion measuring 11.5 cm × 10.5 cm causing destruction of the L5 vertebra and sacrum with replacement of spinal and sacral canal contents. Left iliac bone was destroyed posteriorly. Ano-rectum was displaced laterally, and the bladder and uterus compressed anteroinferiorly. L5-S1 disc showed partial involvement posteriorly by the mass lesion. A possible diagnosis of chordoma was suggested. The patient was referred for fine needle aspiration (FNA) cytology. The aspirate was mucinous. Slides were air-dried and fixed in alcohol and stained by Giemsa and Papanicolaou stain, respectively. Smears were highly cellular displaying a papillary pattern with fibrovascular cores; some clusters of cells were arranged in pseudorosettes with a central myxoid to hyaline area surrounded by a radial layer of cuboidal to columnar cells. Hyaline globules, fibrillary cytoplasmic structures, and myxoid material were also seen [Figures –]. The nuclei of these cells were round to oval with speckled chromatin. Intranuclear inclusions, nuclear grooves, cytologic atypia or mitotic activity was not evident.
Based on these cytomorphologic features, a possibility of MPE was suggested, and the patient was taken up for surgery. The tumor, although adherent to several surrounding structures was resected. Histopathological examination confirmed the FNA diagnosis of MPE and demonstrated a branching papillary pattern characteristic of the tumor. Intercellular myxoid substance was present often surrounding central capillaries. The tumor was immunoreactive for vimentin and glial fibrillary acidic protein (GFAP) and negative for cytokeratin, excluding the possibility of chordoma as was suspected clinically and radiologically.


A 21-year-old-male was referred to our hospital with complaints of insidious onset, gradually progressive pain in the right inguinal region for the last 1-month along with low grade, intermittent fever with evening rise in temperature for the last 2 weeks. The patient had anorexia and experienced significant weight loss. For these complaints, he was investigated in a nearby hospital. Ultrasound examination of abdomen revealed enlarged mesenteric lymph nodes. Fine-needle aspiration cytology (FNAC) of these lymph nodes showed scant lymphoid tissue. He was given a trial of intravenous antibiotics without much improvement. Subsequently, ATT was started.
At the time of the initial examination at our hospital, the patient was febrile and had mild tachycardia and pallor. Patient walked with a limp, and there was flexion deformity at the right hip joint along with fullness in the upper thigh. There was no discrete swelling. Abdominal examination revealed guarding and tenderness in the right iliac fossa. There was no organomegaly or palpable lump. Other systemic examination was unremarkable. Investigation reports showed mild anemia, neutrophilic leukocytosis, very high erythrocyte sedimentation rate (more than 100 mm/1st h), mildly elevated alkaline phosphatase and hypoalbuminemia. Other investigation reports were noncontributory.
Based on history, clinical examination and investigations, provisional diagnosis of psoas abscess with possible tubercular etiology was considered. ATT was continued and intravenous antibiotics were started for the probable secondary infection as evidenced by a neutrophilic leukocytosis. Contrast-enhanced computed tomography (CECT) revealed bulky right ilio-psoas muscles with an ill-defined retroperitoneal (RP) soft tissue mass, medial to the psoas muscle and abutting the right common iliac vessels []. No fluid collection was seen. The possibilities considered at this stage were TB (iliopsoas myositis) and lymphoma. Magnetic resonance imaging of spine and lower abdomen showed infiltrative involvement of iliopsoas and homogeneously enlarged iliac lymph nodes [] that encased right lower ureter, causing moderate hydro-ureteronephrosis. There was no evidence of spondylodiscitis or abscess formation. The differential diagnoses considered were granulomatous (TB, histoplasmosis) disease (with myositis and lymphadenitis) or malignancy (lymphoma).
An ultrasound guided FNAC from the involved muscles was done but was inconclusive. Since the enlarged lymph nodes were close to vessels and the patient was sick, biopsy was postponed and it was decided to continue ATT based on clinico-radiological features, under close, follow-up. During next 2 weeks of hospital stay, the patient showed improvement in terms of response of fever and decrease in total leucocyte count. He was discharged on category I ATT as per DOTS recommendations and advised to follow-up after 2 weeks.
However, patient presented to the medical OPD within 10 days of discharge with complaints of increasing abdominal pain, vomiting, and jaundice for last 5 days. On abdominal examination, diffuse guarding and tenderness with sluggish bowel sounds were noted. Investigations revealed mild anemia, marked neutrophilic leukocytosis and direct billirubinemia with elevated levels of transaminases. An erect abdominal radiograph was normal. CECT abdomen showed increase in the bulkiness of right iliopsoas muscle, increased size of RP soft tissue mass and iliac lymph nodes. In addition, there were multiple mesenteric soft tissue masses/lymph nodes with contiguous involvement of the wall of second and third parts of the duodenum, and moderate ascites []. Considering the rapid progression of the disease, possibility of lymphoreticular malignancy with sub-acute intestinal obstruction was considered highly probable. The patient was managed conservatively with intravenous fluids, antibiotics, and continuous nasogastric aspiration. Upper gastrointestinal endoscopy showed edematous mucosa with areas of ulceration, induration, and luminal narrowing at third segment of duodenum. Multiple duodenal punch biopsies were taken. His condition deteriorated, with increasing abdominal distension, sepsis, and septic shock and he succumbed to his illness on 10th day of hospitalization. As no definitive diagnosis had been arrived at till that time, postmortem liver and lymph node biopsies were performed.
Histopathological examination of duodenal biopsy [] showed multiple mucosal fragments with a dense infiltrate in the lamina propria. Histomorphological and immunohistochemical features suggested a diagnosis of peripheral T-cell lymphoma, not otherwise specified (PTCL, NOS). Liver biopsy showed infiltration by a neoplasm with similar histomorphological and immunohistochemical features.


A 59 year-old male patient suffered from hypertension and dyslipidemia. He was a smoker
and had a positive family history for coronary artery disease. Following an acute
myocardial infarction in February 2013, he underwent a circumflex coronary stent
implantation. Twenty-five days after stent implantation, the patient presented with
acute coronary symptoms, which were found to be due to stent occlusion; however, another
angioplasty proved to be impossible due to technical difficulties.
In August 2013, the patient suffered heart failure, functional class III (NYHA). A giant
aneurysm of the left ventricle was present. Transesophageal echocardiography (TEE) and
cardiac magnetic resonance imaging (MRI) were performed (ejection fraction: 19%
[Simpson]; left-end diastolic volume: 402.7 cm3; left-end systolic volume:
324 cm3; ejection fraction: 19%; left-end diastolic volume: 490 ml; left-end
systolic volume: 398 ml). Left ventricle weight was 144 gm2. The aneurysm was
7.3 x 6.4 x 7.5 cm with tapered walls towards the base of the left ventricle; a thrombus
was present ( and ).
The patient underwent repair surgery of the left ventricle with geometric correction
through a median sternotomy (video 1). Cardiopulmonary bypass (CPB) from the aorta to
the right atrium was established under normothermia (). Myocardial protection was held with anterograde and retrograde
cardioplegia under continuous normothermic esmolol, potassium, and magnesium.
After incising the aneurysm (video 2) and extracting a large thrombus (video 3)
measuring 8 x 3 cm (), a 7 x 5 cm bovine
pericardial patch was placed and anchored with Teflon wires (videos 4 and 5). A
transition zone was established between the healthy myocardium and an area of fibrosis
(video 6) using 2.0 ethibond thereby excluding the infarcted region and a geometric
correction was performed (). The mitral
valve was competent. Cardiopulmonary bypass time was 56 minutes and the aorta was
clamped for 48 minutes. The patient was weaned from the CPB with a low dose of
intravenous dobutamine, which was maintained until closure of the incision.
A new transesophageal echocardiography was performed and revealed a 30% (Simpson)
ejection fraction; left-end diastolic volume of 138.6 cm3, and left-end
systolic volume of 96.87 cm3 ().
The patient was extubated in the operating room and transferred to the intensive care
unit where he remained for 36 hours. Intraoperative blood loss was 450 ml. He was
discharged 72 hours later with prescriptions for carvedilol 12.5 mg daily and
acetylsalicylic acid 100 mg daily. At the one month follow-up examination, the patient
was at functional class I (NYHA). He underwent an MRI that identified: ejection fraction
of 41%, left-end diastolic volume of 198 ml, left ventricular systolic volume of 115 ml,
and left ventricular weight of 144 gm2 ().


A 48-year-old woman presented within 3h after having been bitten on the lateral border of right foot by an Echis carinatus. At the local hospital, management consisted of antibiotics, antitetanus prophylaxis, analgesics, and 10 vials of polyvalent antisnake venom (ASV). ASV was repeated on the next 2 days. On the 3rd day, patient felt dull aching pain in the right flank, hence she was referred to us. She also had breathlessness and heaviness of right chest. She had no neurological deficit. She denied for history of trauma, hematuria, hemoptysis, hematemesis, and bleeding from any other site. Patient was not suffering from any chronic illness and had no addiction. On examination she had swelling of the right foot not extending beyond ankle joint. The blood pressure was 110/70 mmHg, pulse rate was 78 beats/min, and respiratory rate was 20 breaths/min. There was decreased air entry with dull note on percussion on lower zone of right lung. Cardiac auscultation was normal. The whole blood clotting time (WBCT) was > 20 min. CBC revealed hemoglobin (Hb) of 6.4 g/dL, total leukocyte count of 12,000/mm3, with normal differential count. Platelets were 214,000/mm3. Renal and liver functions were normal. Serum sodium was 138 meq/L and potassium 3.5 meq/L. Urinalysis was normal. Coagulation profile showed bleeding time (BT) 3.5 min (normal up to 5 min), clotting time (CT) 12.25 min (normal up to 8 min), prothrombin time (PT) 20 s (control 13 s) with international normalized ratio (INR) of 1.6, thromboplastin time with kaolin (PTTK) 40 s (control 35 s), and fibrinogen degradation product (FDP) was 1.3 μg/mL (0–1). Chest X-ray posteroanterior (PA) view showed moderate amount of right pleural effusion []. For the evaluation of right flank pain ultrasonography was performed which showed hematoma of right adrenal gland. Echocardiography was normal. One liter of pleural fluid was drained which was hemorrhagic. Contrast-enhanced computed tomographywhole abdomen was done on 6th day of being bitten which showed lobulated altered signal intensity in the right adrenal gland measuring 90 × 53 × 31 mm, suggestive of adrenal hematoma [Figures and ]. Computed tomography also showed moderate amount of right pleural effusion []. Two unit of packed cell and six bags of fresh frozen plasma (FFP) were transfused. The pain in the right flank started subsiding and by the end of 2nd week the patient was asymptomatic and had normal hematological parameters.


A 51-year-old male patient was referred to Asan Medical Center on August 10, 2005 for a rectal NET that was found incidentally on screening colonoscopy. The patient had undergone hemorrhoidectomy in his twenties, but otherwise had no significant past medical history. He was a non-smoker and non-drinker. His height and weight were 164 cm and 59.3 kg, respectively. His initial vital signs included a blood pressure of 116/74 mmHg, pulse of 84 beats/minute, respiratory rate of 20 breaths/minute, and body temperature of 36.6℃.
Physical examination revealed no abnormalities. Laboratory data and the results of urinalysis and stool examination were all within normal limits. Chest and abdominal radiography were also unremarkable. Colonoscopy revealed a 5-mm-sized subepithelial tumor in the rectum, 5 cm above the anal verge; histological analysis of a forceps biopsy specimen revealed a rectal NET that was positive for both synaptophysin and chromogranin. Abdominopelvic CT showed no definite rectal lesions, but did reveal a 7-mm-sized perirectal lymph node. Considering the small size of the lymph node and the rare incidence of small metastatic rectal NETs, the malignant potential of the perirectal lymph node was regarded as very low. We discussed the CT findings with the patient, and finally decided to first remove the primary rectal NET endoscopically, with more invasive surgery to be considered if histological analysis revealed any other unfavorable findings, such as lymphovascular invasion, ≥2 mitoses per 50 high-power fields, and/or Ki-67 index >2%.
Endoscopic mucosal resection was performed (). The tumor measured 8×5×5 mm, and the vertical resection margin was positive. The tumor involved the mucosa and submucosa without angioinvasion. The Ki-67 index was 0.8%. Hence, the lesion was diagnosed as a well-differentiated grade 1 NET (). Given the favorable histological results of the resected specimen, we considered metastasis to the perirectal lymph nodes to be extremely unlikely; thus, we elected to follow-up the patient using endoscopy (annual sigmoidoscopy for the first 3 years, then colonoscopy every 3 years) and annual abdominopelvic CT.
For up to 7 years after endoscopic resection, the size of the perirectal lymph node remained unchanged on CT, and no intraluminal recurrence was identified on serial endoscopic follow-up examinations. However, 7 years after the resection, the lymph node was found to have enlarged slightly, from 7 to 10 mm (), although colonoscopy revealed no evidence of recurrence in the rectum. Transrectal ultrasound-guided biopsy was performed on the perirectal lymph node, and histological analysis revealed a well-differentiated NET. Preoperative MRI showed two perirectal lymph nodes suspicious for metastasis. Laparoscopic low anterior resection and lymph node dissection were also performed. The surgical specimen contained two metastatic lymph nodes, measuring 10×8×8 mm and 6×5×5 mm, respectively. Although lymphovascular and perineural invasion were present, the resection margins were clear. The specimen was positive for synaptophysin, but negative for chromogranin. The Ki-67 labeling index was ≤2%, but no mitosis was noted. Finally, the surgical specimen was diagnosed as a metastatic, well-differentiated, grade 1 NET (). The patient was discharged without complications and was scheduled to continue to receive follow-up examinations on a regular basis.


A 22-year-old male patient visited the outpatient department of otorhinolaryngology with a 6-month history of persistent difficulty swallowing after overusing his voice. The patient complained mainly of difficulty swallowing, pharyngolaryngeal pain, throat discomfort, and voice changes. He also complained of intermittent aspiration and dyspnea. He was a student teacher. He had no underlying diseases such as myopathy or motor neuron disease, and family history revealed no remarkable abnormalities. But there is a history of traumatic uvular partial amputation at a young age.
Lip and tongue movement, gag reflex, laryngeal elevation was intact, and there was no tongue deviation on physical examination. To identify a possible cause, a laryngoscopic examination was performed. Only hyperemia of both the palatine tonsil and posterior wall of the pharynx was observed by laryngoscopy (). The auditory perceptual voice analysis was performed to evaluate changes in the voice. An abnormally strained choked voice and voice break were observed. As for the vocalization type, it was a grade 1 (breathiness score 1 and strain score 1) on the scale of Grade Roughness Breathiness Asthenia Strain (GRBAS), so spasmodic dysphonia was suspected [].
He was referred to the outpatient department of rehabilitation medicine for evaluation and a videofluoroscopic swallowing study. The deep tendon reflexes in all 4 extremities were normal. An electrodiagnostic study, including laryngeal electromyography was executed to rule out myotonic dystrophy. There were no abnormal findings on the electrodiagnostic study, and laryngeal electromyography had a normal pattern of muscle activation.
The next day, a videofluoroscopic swallowing study was carried out according to the modified Logemann method []. Parameters representing oropharyngeal swallowing function were analyzed while swallowing thin and thick barium liquid (). There was no penetration or aspiration on thick (200%, BaSO4) and thin (70% BaSO4) liquid swallowing. Oral transit time, pharyngeal delay time, and pharyngeal transit time were all within the normal range, but there was minimal pharyngeal residue on the vallecula area, and twitch-like involuntary movement on the laryngeal muscle around the vocal folds (). He did not feel this involuntary movement, which occurred before and after swallowing.
Based on the results from the VFSS findings, voice aspect, voice analysis, and physical examination, the patient was diagnosed with spasmodic dysphonia. He was scheduled for to be injected with botulinum toxin, but he did not have it done because of business obligations. He recently reported via telephone interview that there was an improvement in his voice, but the voice change reappears when he gets fatigue or speaks too much. Also, symptoms of aspiration, difficulty swallowing, and a foreign body sensation in the throat were still present.


This case was reported to the adverse drug event manager of the Capital Region of Denmark.; A 65-year-old man with type II diabetes, mild hypertension and a previous history of gout was admitted to the Bispebjerg Hospital, University of Copenhagen, Denmark. The patient's daily prescriptions were indapamide 1.5 mg, allopurinol 300 mg, metformin 2000 mg and simvastatin 40 mg.
Fourteen days prior to hospitalization, he experienced muscle fatigue and soreness in the lower extremities. The symptoms gradually progressed to the upper limbs and neck. The general practitioner suspected polymyalgia rheumatica. On arrival, his blood pressure was 153/88 mm Hg, his pulse was 87 bpm and his temperature was 37°C. The blood samples revealed rhabdomyolysis, severe hypokalemia and normal renal function []. Furthermore, the anamnesis uncovered a newly acquired habit of a relatively high consumption of licorice (approximately 100 g/day). There were no signs or history of trauma, diarrhea, polydipsia, infection, intoxication or alcohol intake.
Simvastatin and indapamide were discontinued. Treatment with IV fluids, including potassium substitution, returned the patient's potassium level to normal within 2 days and creatine kinase gradually decreased []. The patient was discharged after 9 days of hospitalization with no signs of kidney injury and an estimated glomerular filtration rate above 90 mL/min.


A 60-year-old man with a medical history significant only for an anxiety disorder presented to a community hospital in Winnipeg, Manitoba with a four-day history of increasing fatigue, weakness, cough and shortness of breath. The patient also complained of sharp right-sided pleuritic chest pain, as well as right upper quadrant abdominal pain associated with deep breathing. He denied nausea and vomiting. A review of systems was otherwise negative. The patient was a heavy smoker and reported occasional alcohol consumption. He denied the use of intravenous recreational drugs.
On physical examination at the time of presentation, the patient was noted to be febrile, with an oral temperature of 39.2°C. He was otherwise hemodynamically stable. The patient was edentulous. On respiratory examination, decreased air entry was noted in the right lower and middle lobes. Bronchial breath sounds were noted at the right lung base, and crepitations were heard with auscultation over the rest of the right lung. The abdomen was tender to palpation in the right upper quadrant. The remainder of the physical examination was unremarkable. Laboratory investigations performed on presentation demonstrated an elevated total leukocyte count (17.8×109 cells/L) with a neutrophil predominance (neutrophil count 16.12×109 cells/L). Blood cultures (aerobic and anaerobic) were negative. On chest radiography, a right-sided pneumothorax with right lower lobe consolidation and associated pleural fluid was apparent (). The patient underwent a noncontrast computed tomography (CT) scan of the abdomen to rule out a subdiaphragmatic abscess in view of the right upper quadrant abdominal pain. No intra-abdominal abnormality was noted; however, the assessment did confirm a right-sided hydropneumothorax, with areas of pulmonary consolidation and loculated pleural fluid.
The patient was admitted to hospital. A chest tube was placed, and empirical antimicrobial therapy with a combination of levofloxacin and metronidazole was initiated. A sample of pleural fluid was submitted to the microbiology laboratory for further analysis. No polymorphonuclear cells or bacteria were observed on Gram stain. The aerobic culture did not demonstrate any bacterial growth following 72 h of incubation. However, after 48 h of incubation, a large Gram-positive spore-forming bacillus was recovered on anaerobic culture. The organism was subsequently identified as Clostridium bifermentans using a Vitek™ 2 ANC card (bioMérieux, Canada). Antimicrobial susceptibility testing was performed by E-test (bioMérieux, Canada), with minimum inhibitory concentrations interpreted according to 2012 Clinical and Laboratory Standards Institute breakpoints for anaerobic bacteria (). The isolate was susceptible to amoxicillin-clavulanate, cefoxitin, clindamycin, meropenem, metronidazole and penicillin.
On the third day postadmission, the patient continued to complain of significant dyspnea and chest pain. A CT scan of the chest was performed with contrast and this demonstrated a right main pulmonary artery embolus. The patient clinically deteriorated and ultimately required admission to the intensive care unit. His antimicrobial therapy was changed to a combination of piperacillin-tazobactam and levofloxacin, and heparin was initiated as treatment for the pulmonary embolus. Fifteen days postadmission, the patient underwent a thoracotomy and decortication of the right lung. Empyema fluid obtained at the time of surgery was submitted to the microbiology laboratory. C bifermentans was once again recovered on anaerobic culture. Postoperatively, antimicrobial therapy with piperacillin-tazobactam alone was continued.
Following surgery, the patient experienced intermittent low-grade fevers (approximately 38°C) and ongoing hypoxia requiring 2 L of oxygen via nasal prongs. Repeat chest radiography revealed a loculated right-sided pneumothorax with a small amount of fluid remaining in the empyema cavity. Twenty-nine days after the initial surgical procedure (44 days postadmission), the patient was taken back to the operating room for a second thoracotomy, right rib resection and empyema drainage. Lung tissue obtained at the time of this surgery for aerobic and anaerobic bacterial culture was found to be sterile. After the second surgery, the antimicrobial therapy was changed to a combination of ceftriaxone and metronidazole. An echocardiogram was performed, which did not demonstrate any evidence of endocarditis or other valvular pathology. The patient gradually improved over a period of several weeks. He was ultimately discharged home 76 days post-admission with a prescription for oral metronidazole to complete an additional 10 days of therapy as an outpatient (arbitrary treatment duration of six weeks following the final surgical drainage procedure).


A 42-year-old white man was admitted to hospital with a 3-day history of progressive muscle weakness, myalgias and dark urines. He was HIV/hepatitis C virus (HCV) coinfected, with an absolute CD4+ T lymphocyte count of 393 cells/mm3, a serum HIV-1 RNA load of <40 copies/mm3 and an HCV RNA load of 6 log10 before admission to the hospital. His antiretroviral treatment consisted of lamivudine (150 mg bd), abacavir (300 mg bd), indinavir (400 mg bd) and ritonavir (100 mg bd) at the time of admission. This regimen had not been changed in the past 2 years. The patient also had a history of hyperlipidaemia, coronary artery disease and a cerebral stroke in 2004. Because of severe hyperlipidaemia (LDL cholesterol, 175 mg/dL; triglyceride level, 191 mg/dL) that was refractory to dietary therapy and a 6-month trial of pravastatin (40 mg/day), a combination of ezetimibe (10 mg/day) and simvastatin (40 mg/day) was introduced 3 weeks prior to admission. The patient was also taking aspirin (75 mg/day), bisoprolol (5 mg/day) and perindopril (2 mg/day). He took no herbal or over-the-counter medications, and he did not use alcohol or illicit drugs. His basal serum creatinine level was 65 μmol/L (0.81 mg/dL). Calculated creatinine clearance using the modified diet in renal disease formula (MDRD) was 98 mL/min.
Ten days after the initiation of treatment with ezetimibe–simvastatin combination, the patient noted the development of new generalized and progressive muscle weakness and dark urines. He denied having recently exercised strenuously or having sustained a trauma, and he had no prior history of HIV myopathy. He was well nourished (weight 66 kg). Laboratory evaluation revealed findings that were consistent with rhabdomyolysis [creatine kinase (CK), 206 000 U/L with normal troponin-I level; alanine aminotransferase, 4200 U/L; aspartate aminotransferase, 1193 U/L; myoglobinuria, 26 700 μg/L and aldolase, 1990 U/L] without acute renal failure (serum creatinine, 0.68 mg/dL; BUN, 3.05 mmoL/L; MDRD GFR 100 mL/min). The urine was positive for myoglobin. Serum levels of amylase and lipase were within the normal range of 49 U/L (<100) and 22 U/L (7–60), respectively. Albumin level was 3.24 g/dL. Results of blood cultures, toxicology screening tests and serological testing for acute hepatitis and antinuclear antibodies, anti-smooth-muscle antibodies and rheumatoid factor were negative. Electromyography was not performed. No clinical or radiological evidence of active infections was documented.
The lipid-lowering agent was immediately discontinued while all antiretroviral drugs were maintained. The patient was treated with massive hydration and urine alkalinization. Clinical symptoms and laboratory abnormalities gradually improved, and within 10 days the patient fully recovered with normal muscle enzymes. He never developed acute kidney injury: serum creatinine was 43 μmoL/L (0.54 mg/dL), 37 μmoL/L (0.46 mg/dL) and 53 μmoL/L (0.66 mg/dL) 3 days, 7 days and 15 days after admission, respectively.


A 59-year-old woman with end-stage renal failure due to systemic lupus erythematosus was admitted to our unit in October 2003 for her first renal transplantation. Her past history was uneventful. She was a non-smoker.
The donor was a 31-year-old woman and there were five HLA donor–recipient mismatches. Initial immunosuppression consisted of sequential quadruple therapy using induction by antithymocyte globulins followed by steroids, mycophenolate mofetil and tacrolimus (FK). The cytomegalovirus (CMV) status was donor positive and recipient negative. Prophylactic treatment by valacyclovir was then given for the first 3 months post-transplant. Apart from an acute episode of functional renal insufficiency, the post-transplant period was uncomplicated. Baseline creatinine level was 0.95 mg/dl (84 μmol/l).
Fifteen days after valacyclovir withdrawal, the patient developed neutropaenia (1440/mm3) and thrombopaenia (123 000/mm3). At this time, clinical examination was extremely poor with only a slight cough. CMV antigenaemia was highly positive (705/200 000 cells). The diagnosis of CMV infection post-renal transplantation was then established. Treatment by IV ganciclovir was then started and a rapid improvement was noted. White blood cell count increased from 1440/mm3 to 3000/mm3 and platelet count from 128 000/mm3 to 181 000/mm3 in 6 days and CMV antigenaemia decreased to 425/200 000 cells in 4 days.
Seven days after the introduction of IV ganciclovir, the patient developed dyspnoea, a severe dry cough and fever (39°C). Hypoxaemia was noted (SaO2 85%). The clinical examination of the patient showed acute respiratory insufficiency with diffuse crackles. Chest X-rays showed diffuse interstitial patchy and nodular opacities involving all the pulmonary lobes. Chest computed tomography revealed that these opacities were bronchocentric and predominantly located at the peripheral part of the lung and showed small nodules and thickening of the wall of multiple bronchi (Figure ). Laboratory tests revealed an inflammatory syndrome (C reactive protein: 179 mg/l). Lymphocyte count was 3100/mm3. Plasma creatinine level remained normal. The results of the latex test, antinuclear antibodies and antineutrophil cytoplasmic antibodies were all negative. CMV antigenaemia was still decreasing at 152/200 000 cells. Repeated sputum and blood cultures, and urinary test for legionella antigen were negative. Empirical antibiotherapy using ceftriaxone and ofloxacine was then started. This treatment was inefficient. Thus, flexible bronchoscopy and bronchial washing were performed. Cellularity analysis of the bronchoalveolar fluid showed 650 000 cells/ml with 72% of lymphocytes, 8% of neutrophil polynuclears, 13% of macrophages and 7% of unidentified cells. Special staining showed no acid-fast bacilli, Pneumocystis carinii or other microorganisms. However, the polymerase chain reaction (PCR) for CMV was positive in bronchial fluid. Diagnosis of CMV-induced BOOP post-CMV pneumonitis was suspected. To confirm this diagnosis, transbronchial biopsies were performed. Unfortunately, biopsy specimens were not contributive because of their small size and open-lung biopsy was not considered because of the respiratory state of the patient.
Antibiotics were stopped and corticotherapy immediately started at 1 mg/kg per day. The clinical status of the patient then dramatically improved. Apyrexia was obtained in 24 h and normal respiratory function was recovered within 10 days. Inflammatory syndrome disappeared in 10 days and CMV antigenaemia was negative after 22 days of ganciclovir therapy. Steroids were progressively tapered off and stopped after 1 month of treatment. Four years later, no other pulmonary problems have occurred, and the patient has done well.


A 57-year-old male peritoneal dialysis patient with ESRD, based on an autosomal dominant polycystic kidney disease, was admitted to our hospital. He reported anorexia, malaise, vomiting and diarrhoea. He smokes one pack/day and consistently drinks a minimum of four units alcohol per day [whisky (40% alcohol volume) and Duvel®, a strong Belgian beer (8.5% alcohol volume)]. On physical examination, a grey appearance and discrete bilateral pitting oedema were noticed. Elevated serum transaminases and cholestasis parameters were found. Glomerular filtration rate was 7 mL/min. Plasma lactate was 5 mmol/L (normal = 1–2 mmol/L). An elevated plasma osmolality of 310 mOsm/kg was found (normal = 275–295 mOsm/kg) with an osmolal gap of 20 mOsm/kg. In contrast with the apparent high triglyceride value (996 mg/dL), serum lipaemic index was only 10 U, suggesting glycerol interference []. Repeating the analysis with a glycerol-blanked method revealed a glycerolaemia of 89.8 mg/dL (normal < 2 mg/dL) and a glyceroluria of 205.5 mg/dL (normal < 1 mg/dL) (). Glycerol clearance was only 0.9 mL/min.
On a CT scan of the abdomen, a large thrombus was detected at the wall of the abdominal aorta, without occlusion. Hypodense areas along the falciform ligament suggested hepatic ischaemia. During hospitalization, liver parameters and triglyceridaemia slowly recovered.
Enzymatic analysis of triglycerides can suffer from glycerol interference. In blood, free glycerol concentration is ± 1 mg/dL, equivalent to ± 10 mg/dL of triglycerides []. Increased glycerol concentrations can be caused by pre-analytical factors, by the pharmacological use of glycerol and by GKD giving rise to pseudohypertriglyceridaemia []. As symptomatic isolated GKD is characterized by episodic vomiting, oxidation of free fatty acids and presence of ketone bodies were monitored. No hyperketonuria or hyperketonaemia was reported. Serum FFA concentrations were normal. As pseudohypertriglyceridaemia was unknown in the patient’s medical history, an exogenous glycerol source was suspected. Carbohydrate-deficient transferrin showed a moderate elevation of 2.8% (normal < 1.3%), as well as the MCV (102.8 fL; normal = 80.5–99.7 fL).
The glycerol intake of the patient was measured by assessing the triglyceride and glycerol concentrations in alcoholic beverages (). Among the beers tested, Duvel® showed a superior glycerol concentration of 228 mg/dL. No glycerol was detected in whisky.
The patient’s beverages were analysed for high-molecular-weight alcohols (fusel oils) []. No fusel oils in the patient’s serum/urine and in Duvel® were detected. However, we found 11.4 mg/dL isobutanol and 12.4 mg/dL isoamyl alcohol in the consumed whisky.
Compared with blood donors, the patient showed a significantly (median value: 152 U/L) reduced plasma GK activity (25 U/L) [], which suggests an impaired glycerol handling capacity.


In September 2005, a 32-year-old male patient with CF (ΔF508/ΔF508) presented to the Cork Adult Cystic Fibrosis Centre with a 6-month history of peripheral oedema. Past medical history was otherwise unremarkable with infrequent previous infective pulmonary exacerbations and no courses of intravenous antibiotics over the previous 10 years. There was no past history of hypertension, smoking, hearing impairment or diabetes mellitus. Examination showed a blood pressure of 134/82 mmHg and mild pitting oedema. Forced expiratory volume in 1 second (FEV1) % predicted was stable at 57% predicted (2.36 L/min) and serum (creatinine) was 169 μmol/L. Dipstick urinalysis demonstrated 4+ protein without haematuria. A spot urine protein-to-creatinine ratio revealed severe proteinuria at 691 mg/mmol (estimated at 6.1 g protein/24 h) with an associated hypoalbuminaemia (24 g/L). The patient repeatedly had normal oral glucose tolerance tests (OGTT) and normal haemoglobin A1c (all <6.2%). Autoimmune screen was negative. Findings were consistent with a nephrotic syndrome, and treatment with an angiotensin-converting enzyme inhibitor and natriuretic agent was instigated and a renal biopsy performed.
Renal biopsy findings revealed nodular glomerulosclerosis (NGS) (A), identical to the classic Kimmelstiel–Wilson lesion of diabetes mellitus. Congo red staining for amyloid was negative, immunofluorescence was negative for immunoglobulin deposition (polyclonal, IgG, IgA, IgM, C3c) and electron microscopy excluded fibrillary glomerulonephritis, amyloidosis and monoclonal immunoglobulin deposition disease.
Despite standard conservative management, he remained proteinuric, with a progressive deterioration in estimated glomerular filtration rate. An intercurrent growth of a mucoid strain of Pseudomonas aeruginosa led to the initiation of alternate monthly nebulized tobramycin 300 mg bis in die (BID) in July 2007 at his annual assessment, in keeping with best international standards of care for patients with CF. This led to a sharp decline in his renal function, reverting back to a steady baseline rate of renal function decline on discontinuation of same after September 2007 (). No other changes to his therapies were made around this period and he was not on any other nephrotoxic medications. Subsequent OGTTs have all remained normal. It is anticipated that he will require renal replacement therapy over the next 6 months and has been prepared for pre-emptive renal transplantation.


In August 2005, a 25-year-old woman on hemodialysis was referred to our renal clinic with a positive pregnancy test based on serum beta HCG. She had end-stage renal disease secondary to type 1 diabetes and had started dialysis 9 months prior to her visit. Her last menstrual period was 6 weeks prior to the visit. She had had two previous pregnancies, the first resulting in spontaneous abortion in the first trimester and the second resulting in the delivery of a baby at 30 weeks gestation. The second pregnancy was in 2001. She was not told that she had kidney disease during her previous pregnancies. Her past medical history was significant for diabetes since age 9 with retinopathy and nephropathy. She was not aware of any kidney disease until she reached the point of needing dialysis in September 2004 and had been on thrice weekly dialysis since then. Her medications included insulin, RenaGel, PhosLo, folic acid and MVI. On exam, her BP was 112/67 mm Hg and she did not have any edema. Her beta HCG levels were 13 270 mIU/mL at the time of her pregnancy test and increased to 30 232 mIU/mL within 3 days. The plan was to increase her dialysis regimen to more than 20 h per week and she was referred to the care of high-risk obstetrics. She had a transvaginal ultrasound scan, which showed an intra-uterine gestational sac of 5.3 weeks, but no fetal heart tone could be detected. Given that she had HCG levels consistent with 7 or more weeks gestation without visible cardiac activity, she was thought to have a non-viable pregnancy and surgical termination was contemplated once her blood sugars were well controlled. Beta HCG levels were followed with the expectation that they would drop in the setting of a non-viable pregnancy. Her beta HCG levels are charted below:
A transvaginal ultrasound was repeated in 10 days and showed an intra-uterine pregnancy consistent with a gestational age of 7 weeks and with good fetal heart tones at a rate of 128 bpm. She continued with her pregnancy and was started on 5 × weekly dialysis. Although, in the second trimester, she had an abnormal triple antigen screening test for fetal abnormalities, she delivered a healthy baby weighing 3.5 kg at 32 weeks without any further complications.


In 2006, a 56-year-old African-Caribbean male was admitted to our local hospital on Curacao for surgical exploration of a relapsing left-sided submandibular tumour. His medical history was remarkable for relapsing tumours in the head and neck region. The first operation was performed in 1970 when a tumour located below the angle of his right jaw was removed. At that time, no pathological diagnosis was established; Ziehl–Neelsen stain for tuberculosis was negative. He used cocaine intermittently and did not appear for outpatient control. In 2002, he returned with a left-sided elliptic tumour, which had been growing slowly for years. Embolization of a suspected lymphohaemangioma of the neck was performed after angiography. In 2006, he returned again with a relapse on the other side and wanted it removed for cosmetic reasons. On preoperative examination, he had a left-sided, non-tender submandibular tumour of ~ 10 × 7 × 3 cm that was not fixated to subcutaneous layers or epidermis. The blood pressure was 110/90 mmHg. His creatinine was 100 μmol/L, and dipstick urinalysis showed 2 + proteinuria. The tumour could only be removed partially from the submandibular/submental region, and a partial parotidectomy was performed as well. Microscopic examination revealed marked hyperplasia of germinal centres with interstitial fibrosis (A), and extensive infiltration by eosinophils resulting in the formation of eosinophilic abscesses (B). Hyalinized vessels were also seen (A, insert). These findings are compatible with Kimura’s disease ().
Three weeks after surgery, he was readmitted to hospital with a nephrotic syndrome. Since discharge, he had been suffering from progressive oedema of the lower limbs without cardiopulmonary complaints. Physical examination revealed a blood pressure of 140/90 mmHg, ascites and extensive pitting oedema of the lower limbs. Laboratory examination showed a creatinine of 299 μmol/L, urea of 49 mmol/L and albumin of 12 g/L; total cholesterol was 16 mmol/L, high-density lipoprotein (HDL) cholesterol 2.1 mmol/L, low-density lipoprotein (LDL) cholesterol 5.6 mmol/L and triglycerides 1.88 mmol/L. His blood showed a marked eosinophilia of 15% (leucocyte count 5.1 × 109/L) and an IgE titre exceeding 3000 IU/mL (normal < 300). There was proteinuria of 6.1 g/24 h. Renal ultrasound was unremarkable with normal-sized kidneys. Anti-nuclear factor and anti-double-stranded DNA as well as serology and polymerase chain reaction for human immunodeficiency virus (HIV) remained negative. The electrocardiogram was normal. Chest X-ray showed a slightly enlarged heart but no signs of pleural effusion. Light microscopy of a kidney biopsy revealed focal segmental glomerulosclerosis (FSGS) with classic ‘tip lesion’ in 3 out of 10 glomeruli (C). Immunofluorescent immunostaining for IgG, IgA, IgM, kappa and lambda light chains, C3c, and C1q showed only non-specific background staining compatible with heavy proteinuria. Electron microscopy showed diffuse effacement and reactive changes of podocytes (D).
The patient was treated successfully with high doses of furosemide and prednisone 1 mg/kg and lost 11 kg of fluid. After one-half year of follow-up, he remained in remission with a serum creatinine of 90 μmol/L.


A heretofore healthy 18-year-old man presented with a 2-day history of nausea, vomiting and stomach ache while consuming induction (20 g/day for 5 days) and maintenance (1 g/day for the next 6 weeks) dosages of creatine monohydrate for bodybuilding purposes. He did not have any significant past medical or family history. At physical examination, he weighed 74 kg, and his BMI was 24.18 kg/m2. His blood pressure was 150/90 mmHg, and he had abdominal tenderness. The initial laboratory studies were as follows: serum urea 39.98 mmol/L (normal 0–36 mmol/L), serum creatinine 201.55 mmol/L (normal 44.2–106 mmol/L), uric acid 0.37 mmol/L (normal 0–0.33 mmol/L), potassium 3.56 mmol/L (normal 3.5–5.0 mmol/L), sodium 148 mmol/L (normal 136–145 mmol/L), pH 7.36 (normal 7.35–7.45), Hct 36.8 (normal 37–52) and total protein 64.87 g/L (normal 64–87 g/L). Urinalysis revealed only proteinuria, and daily protein excretion was 284 mg. The other biochemical parameters and blood count were normal.
The patient was hospitalized, and the creatine supplements were discontinued. Intravenous fluids were administered. During hospitalization, his serum creatinine level increased to 403.10 mmol/L (). Serology revealed negative antinuclear, anti-double-stranded DNA and anti-neutrophil cytoplasmic antibodies. The spiral computed tomographic scan and ultrasonography revealed no abnormalities in the kidneys.
The renal biopsy revealed focal tubular injury with dilatation of tubular lumina and flattening of the tubular epithelial cells. Some of them had hyperchromatic nuclei and prominent nucleoli with occasional mitotic figures. There were sloughed epithelial cells, leucocytes and cellular debris in the tubular lumina; however, there were no pigmented casts. The glomeruli appeared to be normal. Immune complex deposition was not identified with immunoflourescence staining. With these features, the renal biopsy diagnosed acute tubular necrosis ().
Twenty-five days after stopping the creatine supplements, the patient's blood pressure (120/70 mmHg), serum creatinine (88.4 mmol/L) and proteinuria (82 mg/day) normalized, and the patient was discharged from the hospital with a weight of 72 kg.


We report the case of a 40-year-old man who hurt his right foot a few months before in the tropics. He presented himself to the hospital for chronic abscess of the right foot. His medical history revealed that he has previously undergone renal transplantation and has suffered from tuberculosis. The patient came from Djibouti and was in Belgium for a few days. He presented a right foot with a lesion localised on the first toe which was swollen and excoriated (Figures and ) and was self-treated ineffectively with various local antiseptics for several months. The patient had trouble walking and could not wear shoes because of violent pain. X-ray scans revealed no periosteal reaction. The ultrasound of the right foot showed inflammation involving the first metatarsophalangeal joint and a plantar abscess of 3 × 0.8 × 2.3 cm that extended later to the dorsal part of the joint. Ultrasound examination suggests the diagnosis of mycetoma involving lymph nodes.
An open biopsy was done in the operating room by plantar and dorsal approach. Three nodules in the plantar part and six nodules on the dorsal part were found. The role of lymph nodes dissemination was not excluded.
Histologically, the specimen consisted of inflammatory tissue containing lymphocytes and polymorphonuclear neutrophils. Direct examination and culture revealed fungus, but the precise identification was impossible. The decision to not treat with oral itraconazole was taken because of the favourable evolution of the foot. Pain diminished after a few days, and the patient was able to walk after a few weeks.


A 50-year-old female was diagnosed with Gitelman syndrome at the age of 20 years. She was treated with spironolactone (100 mg/day), magnesium lactate (1.2 g/day), potassium chloride (5.4 g/day) and indometacin (25 mg/day).
The patient was recently admitted to our Nephrology Department with oliguria and progressive facial and lower limb oedema 15 days in duration. Her blood analyses showed urea 60 mg/dL, creatinine 0.93 mg/dL, Na 138 mmol/L, K 3.3 mmol/L, Cl 98 mmol/L, Mg 2.1 mg/dL, Ca 8.2 mg/dL (ionic 1.08), P 3.1 mg/dL, total proteins 4.5 g/dL, albumin 1.7 g/dL, bicarbonate 30.1 mmol/L, cholesterol 441 mg/dL, triglycerides 151 mg/dL, high-density lipoprotein 101 mg/dL, low-density lipoprotein 339 mg/dL. Anti-nuclear antibodies, antineutrophil cytoplasmic antibodies and serologic tests for hepatotropic virus, human immunodeficiency virus and treponema were negative, and her complement system, haemogram and thyroid function were normal. Urinalysis findings were proteinuria 11.7 g/day, Na 74 mmol/L, K 152.6 mmol/L, Ca 5.7 mg/dL (136.8 mg/day) and Mg 3.8 mg/dL. A renal biopsy showed: subendothelial and mesangial segmental C1q deposits, mainly located in the distal areas of the capillary loops (), with discrete mesangial proliferation, slight interstitial fibrosis and hyperplasia of the juxtaglomerular apparatus ().
Her definitive diagnosis was nephrotic syndrome secondary to C1q nephropathy, probably minimal change type. Treatment with steroids at a dose of 1 mg/kg/day was started. The nephrotic syndrome resolved after 6 weeks of treatment.


A 64-year-old previously healthy woman with no history of diabetes or hypertension and with normal renal function was admitted to the hospital for elective resection of a left cavernous sinus meningioma. On post-operative Day 9, she became suddenly tachycardic (heart rate 100–110 beats/minute), tachypneic and developed hypoxemia. Computerized tomography angiogram demonstrated large bilateral pulmonary emboli. She was started on intravenous (i.v.) heparin and an inferior vena cava filter was placed. Blood pressure (BP) transiently dropped to 82/45 mmHg for 13 min, the patient was intubated and phenylepherine and norepinephrine drips were initiated, along with aggressive IV normal saline administration. BP quickly increased but urine output gradually declined and reached a nadir of 274 mL/24 h on PE Day 3 (). IV fluids were continued despite stabilization of BP for unclear reasons. A transthoracic echocardiogram performed on the day following PE showed combined right ventricular pressure and volume overload, with a severely elevated right ventricular systolic pressure of 80–85 mmHg and severe tricuspid regurgitation. A pulmonary artery catheter showed an elevated central venous pressure (CVP) and moderately reduced cardiac index (). Interestingly, despite oliguria, creatinine remained normal as did levels of all other electrolytes. The fractional excretion of sodium (FeNa) was 0.097% and the urine sediment was acellular and without granular casts. Renal ultrasound revealed normal kidney size bilaterally and no hydronephrosis. lists urine output, laboratory values and hemodynamic parameters in the days following the PE.
On PE Day 3, the patient had an elevated jugular venous pressure (CVP 32 mmHg), massive bilateral lower extremity edema extending to the abdominal wall and a distended abdomen with hepatomegaly. Lungs were clear to auscultation and chest radiograph showed small bilateral pleural effusions without interstitial edema. The patient underwent catheter embolectomy and thrombolysis with localized urokinase therapy. The following day, urine output increased to 1343 mL/day. Mechanical ultrafiltration (Aquadex FlexFlow Fluid Removal System; CHF Solutions, Brooklyn Park, MN) was initiated because of massive fluid volume overload, as the patient had received close to 30 L of fluids. Following initiation of ultrafiltration, there was a reduction in CVP, no change in cardiac index and a dramatic increase in urine output (). Repeat echocardiography showed a decrease in right ventricular systolic pressure to 55–60 mmHg and reduction in tricuspid regurgitation. Ultrafiltration was continued for two more days after which there was a minor increase in creatinine that returned to normal within a few days (). The patient remained hemodynamically stable and was discharged with a normal serum creatinine.


A 58-year-old gentleman with a history of poorly controlled hypertension and monoclonal gammopathy of undetermined significance (MGUS) was admitted to our hospital with right lower extremity cellulitis complicated by the hematuria, thrombocytopenia and acute kidney injury (AKI). He initially came to our institution for a consultation on his MGUS and what was described as a recurrent post-infectious GN. Two days after arrival, the patient developed cellulitis and was started on oral cephalexin within 24-h of the initial symptoms. On the next day, he felt no improvement. He began to notice amber-colored urine exactly as it happened in three previous episodes that resulted in acute renal failure and he was later diagnosed as having recurrent post-infectious GN. This was his fourth episode (E4) of the recurrent GN, which prompted his admission to the hospital.
The patient suspected that his initial episode could have happened 8 years earlier after he went on a hunting trip and developed a viral syndrome. Within several weeks, he gained around 30 pounds and developed significant swelling associated with decreased urine output. He did not notice any hematuria. He was treated with steroids and furosemide, and the episode resolved. Nothing happened until 6 years later when he had his first (E1) of his many similar episodes. He developed a sore throat with high fever that was followed by gross hematuria, acute renal failure and profound thrombocytopenia (9 × 109/L) in 1–3 days. A renal biopsy was performed which showed changes consistent with post-infectious GN and acute tubular necrosis (). He was treated with steroids, antibiotics and required seven hemodialysis (HD) treatments before his kidney function recovered. His serum creatinine normalized. Next, a second episode (E2) occurred almost 24 months later with a very similar presentation: sore throat and high fever followed in 1–3 days by gross hematuria, acute renal failure and profound thrombocytopenia (10 × 109/L). He underwent a kidney biopsy which again found features of post-infectious GN and acute tubular necrosis (). This time he was treated with antibiotics, but no steroids. He required 10 HD treatments and ultimately completely recovered his kidney function. His third episode (E3) started 8 months later with exactly the same presentation and course. He was treated with antibiotics, but no steroids and required two HD treatments. The lowest platelets count was 19 × 109/L. Nine days after, the last HD patient presented to our institution for a second opinion. His kidney function was recovering but he still had an elevated serum creatinine at 2.6 mg/dL (230 mcmol/L). His platelet count, other hematologic parameters and complement levels were normal. It was interesting to note that blood cultures and other extensive infectious disease studies were negative for identifiable infections in all of the previously described episodes. Complement (both C3 and C4) level was low in each case with otherwise negative serum serological studies.
On presentation, the patient had a platelet count of 468 × 108/mL and a creatinine of 2.6 mg/dL (230 mcmol/L) (). A week later, while in the hospital, his platelet count had dropped to 40 × 109/L, and his creatinine was up to 3.2 mg/dL (283 mcmol/L). Eventually, his creatinine reached 6.1 mg/dL (539 mcmol/L) and he developed nephrotic-range proteinuria with a urine protein/osmolarity ratio of 7.46 [(mg × kg H2O)/(mOsm × L)]. He was treated with high-dose intravenous (IV) steroids, which lead to resolution of the hematuria and improvement of the renal function and proteinuria. At admission, the patient’s antibiotic regimen was switched to vancomycin which improved the cellulitis symptoms. He was dismissed with the creatinine of 4.6 mg/dL (407 mcmol/L) and by Day 10 of the episode, his creatinine was down to 2.2 mg/dL (194 mcmol/L). The platelet count at that time was 620 × 109/L.
Extensive workup for possible infections turned out negative: blood cultures × 2, urine culture, rickettsial panel, human immunodeficiency virus, hepatitis panel, Babesia, Antistreptolysin O titer (ASO), Ehrlichia, Escherichia
coli, Lyme screen. Dengue fever titers were normal. Malaria smear was negative. Serum serology including anti-nuclear antibodies panel, antineutrophil cytoplasmic antibodies, anti-myeloperoxidase, anti-glomerular basement membrane (GBM), anti strep-O, anti-DNAse, kappa and lambda free light chains, cryoglobulin (at body temperature), proteinase 3, cyclic citrullinated peptide Ab were all negative. Serum protein electrophoresis shows a very small M-spike of 2 g/L in the gamma region. Immunofixation shows a monoclonal IgG kappa. Immunological workup revealed a low IgA (0.26 g/L), IgM (0.38 g/L), IgG (3.46 g/L), IgD (<0.01 g/L) as well as low total complement, C3 (0.32 g/L) and C4 (<0.06 g/L). Hematological workup was negative for thrombotic thrombocytopenic purpura (TTP): ADAMTS13 assay, anti-platelet Ab, coagulation study panel and serum lactate dehydrogenase level were all normal. No schistocytes or other features of hemolysis were appreciated on the peripheral smear. A bone marrow aspirate and biopsy revealed slightly hypercellular bone marrow (60% cellularity) with moderate megakaryocytic hyperplasia. No morphologic features of infection or lymphoma were identified. However, there were markedly increased iron stores.
At dismissal, he was placed on penicillin prophylactically although this was not effective at preventing these episodes from recurring. Two weeks after discharge and 3 days after completion of the steroid taper, the patient developed another (fifth, E5) episode of the GN with very same presentation. Again, he was started on high-dose IV steroids with quick taper leading to resolution of the hematuria and improvement of the AKI and thrombocytopenia. Hypocomplementemia improved in between the episodes. Two weeks later, on the last day of the steroid taper, the patient suffered another (sixth, E6) bout of the GN. This was again alleviated by high-dose steroids with a slow taper. Four weeks later, while on prednisone at 17.5 mg daily, the patient developed another (seventh, E7) episode of hematuria with elevated creatinine and profound thrombocytopenia. Interestingly, the patient recalled that these episodes were usually preceded by nasal stuffiness. Also, he would get a sore throat and sometimes lymphadenopathy in the cervical chains. He had been cultured many times and no organism had ever been identified. He recalled having periodic fevers when he was a child. These episodes usually occurred in the summer time and the fever would come ‘out of the blue’. He would get over them without any lingering effects. The patient has two brothers with ataxia, and one may have had periodic fevers as well. His brothers were, however, diagnosed with Friedreich’s ataxia. He is of French and Spanish descent. After the E7 within 4 months, the patient had two more episodes (E8 and E9) while on steroids.
After E9, we decided to treat him with rituximab (four doses of 375 mg/m2). Incidentally, the patient developed pancytopenia after his rituximab infusion. He was treated with growth factors and blood counts recovered. Within a month of completing rituximab, he had two more episodes (E10 and E11) of GN with characteristic symptoms: urinary tract infection symptoms with high fever, followed in 1–3 days by hematuria, AKI and thrombocytopenia. These two episodes were the only two where his creatinine stayed <2 mg/dL and both episodes lasted <5 days. These were shorter than usual and suggested less severe manifestation of the disease. Both episodes occurred while he was on steroids and the dose was increased and slowly tapered. He had had a total of 11 episodes within the preceding 17 months. At which point, he was started on anakinra (100 mg subcutaneously every other day). With anakinra, he was able to completely taper off steroids. Since then, he had had several episodes of infections, but no more GN symptoms. We followed him for another 18 months and he appears to be disease free. There have been no adverse reactions to anakinra. The patient initially lost 25 pounds. Most of this was due to water weight, which was associated with an improvement of the peripheral edema. The weight loss eventually stabilized, and he has not lost any more weight for the last few months. The patient denies any fever. His kidney function had been stable with a normal serum creatinine, no proteinuria and normal urinary sediment. His energy level remained good and he returned to work full time.


A 57-year-old male patient presented with complaints of right upper limb focal seizures since 2 months, speech difficulty of 1-month and 20 days of bifrontal headache and right upper limb weakness. On examination, he was conscious, oriented, dysphasic and had left facial upper motor neuron paresis with right arm weakness of 4/5 power. Magnetic resonance imaging of the brain revealed a left insular irregular enhancing heterogenous lesion with mass effect []. Our provisional diagnosis was high-grade glioma. He underwent left frontotemporal craniotomy, excision of the lesion and discharged. On the 8th postoperative day, he presented with altered sensorium and worsening of hemiparesis. Computed tomography scan revealed a large temporal operated site extracalvarial and extradural hematoma (EDH) with midline shift []. Re-exploration and decompression were done. He deteriorated again on the 2nd postoperative day and had to again re-explore. Surprisingly, he found to have a recurrence of hematoma on the postoperative scan. Since we were quite sure during the third time that adequate hemostasis care was taken, we thought of the other possibilities for the cause and considered angiogram, which revealed an aneurysm along the parietal branch of STA. Hence, he was re-explored for the fourth time and underwent ligation and excision of the aneurysm along with evacuation of EDH.
Histopathology was reported as Granuloma, and the intracranial biopsy revealed Tuberculoma. It was a pseudoaneurysm since there was no true wall noted in the anueysm specimen. There were features of inflammation in the form of lymphocytes and histiocytes. He was treated with antitubercular drugs. He has been on regular follow-up since 1-year. He recovered normally and had no focal deficits.


A 53-year-old man with gout and chronic renal disease was admitted to the hospital because of right flank pain, nausea, vomiting, and fever. A 2.0-cm renal stone in the lower calyx and a 0.4-cm stone in the upper ureter without hydronephrosis were identified on computed tomography images (). The patient was treated with antibiotics and underwent ureteroscopic lithotripsy and stent placement to decompress the infected kidney and extracorporeal shock wave lithotripsy (ESWL) of the renal stone (). After the signs of infection subsided and the uropathy improved (changes in high-sensitivity C-reactive protein from 22.2 to 2.75 mg/dL and in creatinine from 3.6 to 1.7 mg/dL), we performed several sessions of ESWL on the right renal stone. Two months later, we found Steinstrasse around the stent (). We attempted to change the stent but failed to remove it because of resistance. We therefore checked for an encrusted stent by x-ray and computed tomography, which revealed a knotted stent and Steinstrasse (). The patient refused general or spinal anesthesia, so the stent was removed percutaneously under local anesthesia.
We inserted a 7-French angio sheath using ultrasonographic and fluoroscopic guidance and then inserted a KMP (Beacon Tip Torcon NB advantage angiographic catheter, Cook Medical, Bloomington, IN, USA) catheter. Finally, a folded 0.018 Terumo guidewire was inserted into the area of knot formation through the KMP catheter. The knotted stent was caught in the folded guidewire and removed percutaneously. Subsequently, an 8.5-French pigtail catheter was inserted in the kidney (). After several sessions of ESWL performed within 2 weeks of the surgical procedure, the remnant stone and Steinstrasse were completely eliminated and the pigtail catheter was removed.


A 23-year-old woman with a history of 1 year of typical trigeminal neuralgia with left-sided lancinating facial pain in the V2 distribution of the trigeminal nerve. The pain was triggered by certain movements, tactile stimulation, or a cold shower. She also complained of neck pain and facial numbness. Physical examination findings are shown in . ( Low-set hairline and brevicollis [shortened neck] presenting as clinical features of craniocervical junction malformations).
The neurologic exam shows decreased left corneal reflex, nystagmus, hyperreflexia, and presence of bilateral Hoffmann's sign. Magnetic resonance imaging (MRI) demonstrated basilar impression. ( Preoperative sagittal T2-weighted magnetic resonance imaging [MRI] showing basilar impression with compression of pons by the tip in the topography of the odontoid apophysis located 12 mm above the Chamberline line and deformity of the posterior fossa with asymmetry of petrous bone).
The operation was performed through a osteodural decompression with large inferior suboccipital craniectomy. After dural opening shaped in “Y,” the arachnoid adhesions between the cerebellar tonsils, medulla oblongata, and spinal cord were dissected. A large opening of the fourth ventricle was obtained and dura was grafted with bovine pericardium. The neuralgia disappeared after surgery and remains completely resolved 1 year later.


63 year old male with history of active heroin abuse, non-compliance, hypertension, obesity, CHF, hypercholesterolemia, multiple prior intraabdominal surgeries, including appendectomy, cholecystectomy, several ventral hernia repairs, long history of large ventral hernia, presented with worsening abdominal pain and distention for 2 weeks. Two days prior to admission, he noticed that his ventral hernia on right side of the abdomen became more swollen and painful. In the ER he became obtunded, developed severe respiratory distress and required endotracheal intubation. Subsequent CXR showed free intraabdominal air. Plain abdominal radiograph showed markedly distended cecum pointing to the left upper quadrant and free air (). At this point surgical consultation was obtained. Vitals: T 99, BP 174/90, pulse 115, respirations 12. Clinical exam showed distended abdomen and very large tender hernia occupying all right side of the abdomen and extending to the left of midline with redness and warm skin above it. Patient had multiple surgical scars and another 2 smaller hernias in lower abdomen. Abnormal laboratory results: WBC 15,000, creatinine 4.5. Subsequent CT of the abdomen and pelvis without contrast showed tremendous amount of free air and markedly distended cecum, partially contained in the ventral hernia and crossing midline to the left, suspicious for cecal volvulus (). Patient was immediately taken to the OR, where diagnosis of cecal volvulus was confirmed. Cecum, terminal ileum, and ascending colon were twisted in axial plane, with concomitant clockwise torsion and grossly perforated hepatic flexure, significant fecal spillage and large abscess cavity along the transverse mesocolon. Size of the hernia was approximately 20 × 30 cm. Hernia contained part of the cecum. Right colon had significant adhesions from prior surgeries, including those inside the hernia sac. Patient had loss of the domain. There was another lower abdominal wall hernia containing old infected mesh and communicating with major right sided abdominal wall hernia defect. The volvulus was reduced in counterclockwise direction. Extensive lysis of adhesions, right hemicolectomy, abdominal washout, explantation of infected mesh was performed. Because of extreme condition of the patient, damage control procedure was chosen and abdomen was temporally closed with negative pressure (VAC) dressing. Patient remained intubated in ICU. After 48 h of resuscitation patient improved: pressors were discontinued and he had good urine output. Patient was taken back the OR. Reopening of recent laparotomy, ileocolostomy anastomosis and ventral hernia repair with biological mesh was performed. Repair of abdominal wall defect was challenging because of the size of the hernia. Despite meticulous dissection, we had 10 × 12 cm defect in the middle, which was not possible to approximate. This area was bridged with one piece of biological mesh. Another bigger piece of mesh (same type) was placed above the bridged area to have 7–8 cm overlapping from the edges of the hernia. Mesh was secured to the fascia with unabsorbable sutures and titanium tackers. Patient had successful recovery. He was extubated 5 days later, started on regular diet. All drains were removed before his discharge to rehabilitation center. A week after his discharge, patient was readmitted with pneumonia, and CT of the abdomen was performed in the ER. CT showed no bowel obstruction and no hernia (). Patient was discharged to home in stable condition and was followed in the clinic without evidence of recurrence during 4 months after surgery.


A 64-year-old woman presented with weakness in both lower limbs, which had gradually progressed over the previous 4 months, and difficulty in walking on admission. She had no sensory dysfunctions or visceral involvements, but had a surgical history (the removal of thoracic spinal tumor) for a similar episode of bilateral lower leg weakness 16 years ago ().
Clinical examination revealed a tender linear scar extending from the C7 to T9 spinous process. Neurological assessment revealed bilaterally increased muscle tone, associated with symmetrical paraparesis (3/5). No sensory loss or bladder/bowel involvement was evident. Magnetic resonance images revealed at Tl and T2; Godolinium enhanced image showed a well-enhanced tumor mass on the left side in the spinal canal and marked the compression of the spinal cord combined with compressive myelopathy ().
She underwent a second surgery at the same site and a pale yellowish tumor was excised. The pathological examination confirmed that it was psammomatous meningioma (), compared with previously transitional type (microscopic examination showed nests or concentric whorls of menigothelilal cells revealing polygonal or oval cells with eosinophilic and indistinct bordered cytoplasm and round nuclei, also showing of frequent psammoma bodies in the center of the tumor whorls). The patient showed neurological recovery after the operation. Postoperatively, the patient's sensory parameters and bladder control improved. After 2 weeks, she showed a slight improvement of motor power and was discharged.


A 26 year old male patient was changing his car tyre on the roadside when he was hit by another car from the side. On arrival to the emergency department (ED), he was hemodynamically stable with heart rate of 100/minute, BP 100/70 mm of Hg and respiratory rate of 18/minute. He was managed as per Advanced Trauma Life Support (ATLS) protocol. The patient was oriented with negative focused assessment with sonography for trauma (FAST) and inability to lift left leg with pain in the pelvic region. On examination, pelvic compression test was positive. He was having foot drop on the left side. After initial clinical assessment plain anteroposterior radiograph of the pelvis suggested a pelvic fracture. Subsequent computed tomography (CT) scans and radiographs revealed a fracture of the left posterior ilium with anterior dislocation of the SIJ []. There was associated symphyseal disruption with marked widening and high oblique fracture of the left superior ramus.
The patient was operated for his pelvic injuries on the 5th day following trauma. Pfannenstiel incision and lateral window of the ilioinguinal approach were used to access the fractures. The fractured ilium was found to be lying anterior to the sacrum. A Schanz pin was placed onto the iliac crest to get a proper hold of the iliac blade, which was then levered anteriorly, laterally to its anatomical position. Proper care was taken to avoid further damage to the intact yet contused L5 nerve root while reducing the anteriorly placed fractured iliac blade. The fractured fragment was reduced anatomically and fixed with a reconstruction plate (4 hole) across the SIJ in the standard fashion. The symphysis was also stabilized with a 3.5 mm plate and screws []. The total operative time was 2 hrs with a blood loss of 300 ml. Patient was allowed full weight bearing after 8 weeks. At the end of 18 months, patient was back to his pretrauma activities. The functional outcome of the patient was good with recovery of the peroneal nerve function at the last followup at 2 years.


A healthy 28 year old man who was hit by a heavy vehicle while getting down from a moving bus. Patient was hemodynamically stable on arrival in the ED. He was unable to lift both the lower limbs due to severe pain. There was a 5 × 3 cm wound in the left inguinal region. On further radiographic and CT evaluation, there was evidence of a fracture of the left posterior ilium with anterior fracture dislocation of the SIJ with fractured anterior fragment lying in front of sacrum []. Ipsilateral superior and inferior rami fracture were also present. On the right side, there was a typical posterior crescent fracture dislocation with ipsilateral superior and inferior rami fracture.
On the 3rd day, after complete stabilization and work-up, the patient was operated upon for his pelvic injuries. A staged procedure was performed. First, in supine position, the reduction and fixation of the left anterior fracture dislocation of SIJ was performed through the lateral window of the ilioinguinal approach. Application of anterior iliac crest Schantz pin assisted in achieving reduction of the iliac blade. The hip was abducted with the knee extended to aid in reduction by countering the unopposed muscle actions of the abductors of the hip. Total blood loss was 250 ml. After 3 days the patient was operated for the right sided posterior SIJ fracture dislocation in a prone position. Fracture was reduced closed and two lateral compression (LC) II screws from posterior superior iliac spine were placed percutaneously across the crescent fracture under image guidance. The patient recovered well and had excellent functional results at the end of 18 months followup [].


A 36 year old male patient who fell from second floor of his house and presented in the ED with complaints of severe pain in the back and the pelvis. The pelvic compression test was positive. Radiographic and CT scan evaluations [] revealed fracture of the right posterior ilium with a small fragment attached posteriorly to sacrum with anterior dislocation of the remaining ilium in front of sacrum. On the left side, there was a crescent fracture with mild posterior dislocation of the iliac blade.
Associated fractures of superior and inferior pubic rami on the left side and of inferior pubic rami on the right side were also noted. There was no distal neurovascular deficit in both the lower limbs.
Operative intervention was performed on the 4th day following trauma. Both the fractured SIJ were operated upon in the same sitting with standard anterior SIJ plating []. The reduction of the anteriorly placed fractured iliac wing was facilitated by the insertion of the Schantz pin in the supracetabular region to have a proper control of the iliac blade.
Total duration of surgery was 2.5 hrs with a blood loss of <500 ml. The fractures healed completely and the patient was performing all his activities at the last followup at end of 14 months.


A 29 year old male patient who fell from the third floor of an under construction building and was brought to the ED in a haemodynamically unstable position with heart rate of 120/minute, BP 90/60 mm of Hg and respiratory rate of 22/minute. On further assessment it was found that he was having splenic laceration, left renal injury, left ureteric transection distal to pelviureteric junction and an anterior fracture dislocation of the left SIJ along with iliac blade fracture on the right side and dislocation of the left elbow joint []. He had a 7 cm lacerated wound over the left flank. He was urgently managed for his splenic, renal and ureteric injuries. After 7 days, he was operated for his pelvic ring injuries.
Similar maneuver of reduction as described for other cases was performed and the fracture stabilized with two reconstruction plates across the SIJ. The fractured ilium was also fixed with a reconstruction plate (4 hole) and two 3.5 mm cortical screws. Iliac blade fracture on the right side was managed percutaneously with three lag screws []. At 12 months followup, the patient was walking unaided and had no residual pain or instability [].


A 49-year-old man presented with nephrotic syndrome and increased serum creatinine. Past history included hypertension, hypercholesterolaemia, porphyria cutanea tarda and chronic liver disease secondary to hepatitis C virus (HCV) genotype 4. Eight years earlier HCV had been treated with peg-interferon-α plus ribavirin for 13 weeks, but therapy was discontinued due to poor response and poor tolerance. The patient refused further treatment for HCV infection.
A kidney biopsy revealed fibrillary glomerulopathy. He was initially treated with furosemide, thiazide, angiotensin-converting enzyme inhibitor, angiotensin receptor blocker, spironolactone, doxazosin and a calcium channel antagonist. The blood pressure was controlled and proteinuria decreased from 11–15 g/day to 7–9 g/day (11.000–15.000 mg/24 h to 7.000–9.000 mg/24 h). However, serum creatinine rose from 132.6 to 247.5 μmol/L (1.5 to 2.8 mg/dL). Due to progressive impairment of renal function and persistent nephrotic proteinuria, immunosuppressive treatment was started with prednisone (1 mg/kg/day for 1 month and taper) and two 1000 mg doses of RTX separated 15 days.
Five weeks after the last RTX dose, progressive shortness of breath and dry cough developed without fever or other symptoms. He was admitted to the intensive care unit (ICU) because of low oxygen saturation (68%). Computed tomography revealed bilateral interstitial pulmonary infiltrates without pleural effusion. Bronchoscopy and broncho-alveolar lavage showed no bleeding and microbiological studies were negative.
The patient was treated with intravenous antibiotics and high-dose corticosteroids. He was discharged from the ICU after 10 days with the diagnosis of respiratory failure probably due to interstitial pneumonitis secondary to RTX. The serum creatinine level increased to 530.4 μmol/L (6 mg/dL) and renal replacement therapy with haemodialysis was initiated ().


A 37-year-old man visited the emergency room with a left periorbital injury caused by a traffic accident. An initial examination showed no abnormalities in visual acuity, intraocular pressure, or the fundus. The preoperative diagnosis was a blowout fracture involving the medial wall of the left orbit (). Surgical repair of the fracture through insertion of a Medpor (Stryker, Kalamazoo, MI, USA) implant was performed six days after the injury. After the operation, the patient complained of newly developed diplopia. The elevation of the left eye was conspicuously limited during adduction (). A forced duction test of the left eye showed restricted elevation during adduction. A postoperative computed tomography (CT) scan of the orbit showed that the left superior oblique muscle was trapped between the inserted Medpor fragment and the border of the fracture (). Surgical re-exploration was performed and the Medpor implant was removed (). Fortunately, reduction using a bone hook allowed the defect to be almost completely covered with the depressed orbital wall. No artificial bone was inserted. After surgery, a forced duction test showed no restriction of ocular motility. However, the diplopia remained for two months post operatively and was corrected with prism glasses. At an eight-month follow-up visit, the patient's diplopia was alleviated and his ocular motility was significantly improved ().


A 52-year-old man came to the hospital with left periorbital blunt trauma caused by a machine tool. He complained of periorbital ecchymosis and subconjunctival hemorrhage without diplopia and ocular motility limitation. An orbital CT scan revealed a left medial blowout fracture (). Surgical repair with the inser tion of a Medpor implant via a transcaruncular approach was performed five days after the injury. Immediately after the oper ation, he complained of diplopia and limited ocular motility. A forced duction test was positive, demonstrating restriction of el evation during adduction of the left eye. A postoperative CT scan showed entrapment of the superior oblique muscle between the Medpor implant and the orbital wall fracture border (). Surgical re-exploration was promptly performed and the entrapped muscle was released by removing the Medpor implant. A new ly designed Medpor implant was reinserted into the depressed orbital wall (). After three month, the diplopia and ocular motility limitation were completely resolved.


A 52-year-old man was transferred to our department for the surgical repair of a right medial blowout fracture caused by a violent assault. At first, he complained of diplopia in all directions of gaze. An examination showed limited movement in the right eye during abduction. Traumatic hyphema and vitreous hemor rhage were observed. After the hyphema and vitreous hemor rhage were completely stabilized, an operation was performed to correct the blowout fracture. The herniated orbital fat and muscle were restored and a Medpor implant was inserted into the defect of the orbital wall using a transcaruncular approach. After the operation, he complained of aggravated diplopia and limited ocular motility. Limited elevation during adduction of the left eye was also observed. A postoperative CT scan showed that the superior oblique muscle was compressed by the swell ing of adjacent soft tissue and hematoma. Acquired simulated Brown syndrome was strongly suspected. After two months, the patient's diplopia and limited ocular motility had improved and the patient was satisfied with his condition.


An 8-year-old girl was admitted to the pediatric department of the Balaji Hospital, Chrompet, Chennai with the complaints of with high grade fever of 4 days duration associated with nonbilious vomiting, watery stools, abdominal pain, and reduced appetite. Clinical examination revealed hepatosplenomegaly.


A 22-year-old male presented with a 10-year history of asymptomatic and progressive brownish-black discoloration over the dorsa of hands and feet. Pigmentation progressed slowly to involve the dorsal and ventral surfaces of the extremities, neck, and chest. His past medical history was unremarkable. There was no history of photosensitivity. There was no history of handling any chemical directly or any significant history of drug intake. There was no similar case in the family and no history of consanguinity among parents.
Cutaneous examination revealed punctate, irregular, slightly atrophic, brown macules arranged in a reticulate pattern on dorsa of hands and feet, the extensor and flexor surfaces of forearms, flexor aspects of neck and chest []. Brownish pigmentation was also present on the palms with absent dermatoglyphics distal to the distal crease and the fingernails were dystrophic []. Plantar pitting was also present []. Facial skin and teeth were normal. Systemic examination did not reveal any abnormality. Routine investigations including complete blood counts, urinalysis, liver function tests, renal function tests, and serum electrolytes were within normal limits. The Venereal Disease Research Laboratory test and ELISA for HIV were nonreactive.
Skin biopsy was taken from the left forearm, which showed digitate and filiform elongation of rete ridges, with clumps of heavy melanin pigmentation at their tips. There was melanin incontinence in the papillary dermis []. This confirmed the diagnosis of RAK.


A 26-year-old male presented with multiple hypopigmented atrophic macules over the upper and lower limbs since one month []. Dermoscopy revealed whitish structureless areas (WSA), comedo like openings (CLO) [] and dotted vessels arranged in a net-like pattern [].


A 9-year-old girl presented with asymptomatic papules over the face, back, upper and lower limbs since two months. Dermoscopy revealed WSA and telengiectasia [].


A 22-year-old male had atrophic, hypopigmented macules and papules over chest, trunk and lower limbs since 5 months. Dermoscopy revealed WSA, white chrysalis like structures (WCLA) and telangiectasia of different lengths and calibers [Figure and ].
Histopathology of lesions showed atrophy of the epidermis, hyperkeratosis [], and follicular plugging with basal cell degeneration. [] The dermis showed edema and homogenization of collagen and interstitial and perivascular lymphocytic infiltration [].
Hair, nail, oral, genital mucosa, systemic examination, and blood investigations were within normal limits in all three patients. A diagnosis of extragenital LS was made in all three cases based on clinical features and histopathology.


A 50-year-old man was admitted to our hospital owing to early gastric cancer (EGC) detected upon esopha gogastroduodenoscopy (EGD) during a routine check-up. A family history of thyroid cancer was determined, with his sister having undergone resection for thyroid cancer. His medical history included hypertension and, consequently, he received medication for treatment. Physical examination revealed no significant findings. Laboratory investigations did not show any significant abnormalities. The tumor markers were within normal limits. EGD revealed an EGC type IIb lesion at the lower portion of the stomach, and the pathology report showed poorly differentiated adenocarcinoma (). An abdominal/pelvic CT scan showed no significant lymph node (LN) enlargement due to gastric cancer but did show a 1.7-cm exophytic hypervascular renal mass in the inferior pole of the left kidney, which was determined to be renal cell carcinoma ().
We performed thyroid ultrasonography and fine needle aspiration biopsy (FNAB). Two 1.6 and 0.9 cm probably malignant thyroid nodules were found, which was bulging into the superior and lateral capsule with microcalcification, as well as several conglomerated LNs with jugular vein invasion in the right side of the neck (). FNAB results indicated papillary thyroid carcinoma and level IV LN metastasis. 18F-fluorodeoxyglucose (18F-FDG) PET/CT showed a focal FDGavid nodule with calcification in the right upper thyroid lobe, level III and IV LNs metastasis in the right side of the neck, and an isometabolic nodule in the left kidney (). No definite abnormal FDG uptake along the gastric wall was noted.
Our team performed a radical subtotal gastrectomy, a consecutive left partial nephrectomy, and a radical thyroidectomy with modified radical neck dissection in the right side of the neck. Total operative time was 315 minutes and total bleeding amount was check 400 mL.
The pathology report following gastrectomy determined the tumor to be EGC type IIc, tubular adenocarcinoma, moderatelyto-poorly differentiated, intestinal type by the Lauren classification, with a depth of invasion above the muscularis mucosa (T1a) and with no metastases in any of the 45 LNs assessed (T1N0M0; stage 1A). The pathology report following partial nephrectomy showed a 1.9 × 1.5-cm completely encapsulated renal cell carcinoma, a clear cell histology, and a pathologic stage of T1aN0M0; stage 1. And the pathology report following thyroidectomy indicated the presence of a 2.0 × 1.5-cm thyroid papillary carcinoma, with recurrent laryngeal nerve, jugular vein (T4b) and extensive lateral LNs involvement in 10 of 22 LNs assessed (T4aN1bM0; stage 4A). After the three consecutive operations, the patient was in good health and was discharged without any postoperative complications.


A 41-year-old female patient was admitted to our hospital with complains of occasional abdominal pain. She had undergone open cholecystectomy for cholelithiasis fifteen years ago. Physical examination revealed no significant findings apart from previous right subcostal surgical incision. On contrast-enhanced abdominal computed tomography; stages 4 - 5 hydatid cyst, about 8 cm in diameter, accompanied by calcification on subdiaphragmatic surface at segment 7 - 8 of the liver was detected (). The patient underwent laparotomy with a preliminary diagnosis of hydatid cyst. After Adhesions secondary to previous operations were separated, the lesion located between the liver and diaphragm was detected. The lesion was aspirated with a veress needle and then filled with povidone iodine and povidone iodine embedded compress was placed around the lesion. When the lesion was opened, dark yellow abscess content was noticed and aspirated. A surgical compress or towel having partially corrupted fibrous structures was detected within the lesion (). Lesions were 9 - 10 pieces with different sizes. All lesions were completely cleared. A drain was placed before terminating the operation. The drain was pulled out on the third postoperative day. The patient recovered without complications and remained well and symptom-free for at least 8 months after discharge.


A 22-year-old male student, with no significant past history of any medical illness, presented with a history of acute onset hoarseness of voice and binocular diplopia since 10 hours and severe hypophonia with dysphagia since 4 hours. There was no visual blurring or history of eating canned food or abdominal pain or other gastrointestinal symptoms. Patient denied any recent history of fever or any other infection in the preceding weeks to months. There was no prior such episode of bulbar or limb weakness and the current illness did not show any fluctuations in the symptoms. On examination, tachycardia, hypertension and multiple cranial nerve palsies, including bilateral pupil sparing oculomotor, troclear, abducens, trigeminal (including facial sensory impairment), facial, glossopharyngeal, and vagus nerves was detected []. Patient also had generalized limb hyperreflexia but normal tone and power, absent jaw jerk, and flexor plantar reflex bilaterally. Rest examination was unremarkable. No orthostatic hypotension or other signs of autonomic dysfunction were detected. Investigations showed a normal contrast enhanced magnetic resonance imaging (MRI) of brain and computed tomography (CT) of the base of skull. Nerve conduction study (NCS) of facial nerve showed bilaterally decreased amplitude with a neuropathic pattern on electromyography (EMG). Limb NCS and repetitive nerve stimulation (RNS) tests were normal. Ice pack test and neostigmine tests failed to show any improvement in symptoms. Acetylcholine esterase antibody was also negative. Blood sugar, glycosylated hemoglobin, serum electrolytes including potassium level, electrocardiography, and renal and liver function tests were normal. Muscle enzyme levels including creatinine phosphokinase (CPK) and CPK-NAC were normal. Cerebrospinal fluid (CSF) examination done on second day and repeated on eighth day showed normal proteins, two lymphocytes and normal glucose level. Polymerase chain reaction (PCR) of CSF for herpes simplex-1 and varicella zoster was negative. Patient also tested negative for human immunodeficiency virus (HIV), antinuclear antibody (ANA) by immunofluorescence, venereal disease research laboratory (VDRL) antibody, acetylcholine esterase (ACE), and antibodies against borrelia burgdorferi. There was no history suggestive of diphtheria, sarcoidosis, recent ingestion of canned food or treatment with chemotherapeutic agents. The only positive tests were a low TSH level− < 0.01 (normal range = 0.25-5.0 uIU/mL), high free T3 level−19.2 (normal range = 4.0-8.3 pmol/L), and high free T4 level− 39.2 (normal range = 9.0-20.0 pmol/L). Thyroid ultrasonography was normal and anti-thyroid peroxidase (anti-TPO) and anti-thyroglobulin antibodies were negative. Considering the multiple cranial nerve with facial sensory involvement, a diagnosis of polyneuritis cranialis was made. As CSF analysis was normal, a Guillain Barré Syndrome (GBS) variant was considered less likely. In view of abnormal thyroid function tests, a thyrotoxicosis-associated polyneuritis cranialis was concluded and not idiopathic polyneuritis cranialis. Patient was treated with Carbimazole 10 mg thrice daily and Propranolol 40 mg thrice daily, with which over next 2 weeks, he showed partial recovery in jaw muscles, lower facial muscles, dysphagia, and dysarthria, and almost complete recovery in 8 weeks. As neither GBS nor idiopathic polyneuritis cranialis were considered as the final diagnosis, immunomodulatory drugs like immunoglobulins or plasmapheresis, or corticosteroids were not administered.


A 69-year-old male presented to us with a history of droopy skin over the eyelids since the past 2 decades. Being advised to seek medical opinion from elsewhere over a decade ago, he did not undergo any clinical examination because he was neither motivated nor did the lid mass cause him any difficulty except for abnormal cosmesis []. The lesions developed initially as small folds in the skin of the lid [ – left upper lid] and over two decades assumed the current proportions. The fibromas were multiple in number (one on each lid), very slowly progressive, soft and trans-illuminant []. The larger of the masses encompassed the lid fold [] and was pedunculated.
There were no episodes of pain or inflammation. There were no associated findings suggestive of neurofibromatosis. The patient's daughter was getting married in the coming month. It was at her advice and suggestion that the father finally decided to undertake the surgical removal of the lesions. He underwent simple excision [] of the lesions on December 7, 2011 and had an uneventful recovery. The base of lesions was clamped with a small artery forceps and crushed. Surgical scissors were used to excise the crushed area thereby removing the lesion from its base. The availability of loose skin led to overlap of tissue thereby requiring no suturing in layers. Histopathologic examination using hemotoxylin eosin staining revealed stratified squamous epithelium lining a connective tissue stalk showing loose fibro-collagenous tissue containing congested blood vessels and chronic inflammatory infiltrate. No adnexal structures were seen. This confirmed the diagnosis [].


We report a case of a 77-year-old Japanese woman with a history of diabetes mellitus, hyperlipidemia, and abdominal shingles, who presented to her physician with left leg swelling and pain with no other associated symptoms. Upon examination, there was a tender palpable lesion 1.5 cm × 1.0 cm in the anterior aspect of her left leg. Neurological examination was unremarkable. Magnetic resonance imaging of the lower extremities revealed a distal left tibial lesion measuring 11.0 cm × 2.1 cm with focal extraosseous extension [].
Fine-needle aspiration was performed on the patient's left leg mass using a 21 gauge needle. Air-dried smears were prepared and stained on-site with the Diff-Quik stain. Some of these slides were later destained and restained with the Papanicolaou (Pap) stain. The remainder of the specimen was placed in formalin for a cell block preparation. The cytologic smears showed a moderately cellular yield, with the tumor arranged mostly in three-dimensional cohesive clusters with cellular overlapping and architectural disorder []. A few single tumor cells were noted in the background. The tumor assumed the form of predominantly cuboidal to columnar cells exhibiting low nuclear: cytoplasmic (N:C) ratios and partially vacuolated cytoplasm. The cells contained elongated nuclei showing mild to moderate nuclear pleomorphism with finely dispersed chromatin and occasional prominent nucleoli []. No papillary formations or clear cell features were noted. The cell block preparation revealed tumor glands arranged in frequent cribriform structures lined by crowded columnar cells []. Immunohistochemical staining showed diffuse positivity of the neoplastic cells for cytokeratin 7, estrogen receptor, and paired box gene 8 (PAX-8). Staining was negative with cytokeratin 20, CDX-2, thyroid transcription factor-1, napsin-A, gross cystic disease fluid protein-15, and mammaglobin. A diagnosis of metastatic endometrial carcinoma was rendered.
Based on the cytological diagnosis, the patient then underwent systemic work-up including computerized tomography (CT) scans of the abdomen, pelvis, and chest. The abdominal CT showed a 4.2 cm fluid density in the uterus, thought to be within the endometrial canal. A pelvic CT scan, as well as a bone scan, demonstrated a 6.0 cm × 5.2 cm enhancing soft tissue mass destroying the right pubic symphysis. Thoracic imaging revealed bilateral pulmonary nodules measuring up to 4 mm.
Endometrial biopsy performed 2 weeks after the initial FNA revealed endometrioid adenocarcinoma with focal squamous differentiation. Soon thereafter, the patient underwent radiation to the left tibia and pelvis followed by total abdominal hysterectomy with pelvic lymph node dissection. Gross examination showed a 143 g uterus with a tan, firm tumor lesion measuring 4.3 cm × 3.2 cm, occupying the anterior and posterior fundus and body. Cross sections demonstrated tumor invading to a maximal depth of 25 mm out of 26 mm of the myometrial thickness. Microscopic examination confirmed the diagnosis of FIGO grade 2 endometrioid adenocarcinoma, characterized by myoinvasive neoplastic cribriform glands similar to that seen in the patient's bone FNA []. Immunohistochemical staining of the uterine endometrial cancer showed a staining profile similar to that seen in the osseous metastasis, including positivity for cytokeratin 7, estrogen receptor, and PAX-8. No lymph node metastases were detected.
The patient declined radiation or chemotherapeutic treatment, and was started on hormonal therapy with regular follow-up. Two years after her initial FNA diagnosis, she is asymptomatic, and serial bone scans demonstrate therapeutic response of her osseous metastases. A CT scan of the chest, abdomen, and pelvis show no progression of the bony lesions or development of new metastases.