Chylothorax associated with primary membranous nephropathy: a case report.

BACKGROUND: Chylothorax is a state in which pleurisy is induced by chylomicron leakage due to lymphatic injury. Membranous nephropathy (MN) is one of the relatively common glomerular diseases that cause nephrotic syndrome in adults. Chylothorax at the onset of nephrotic syndrome is very rare in adult patients. CASE DESCRIPTION: We report a case of chylothorax associated with primary MN. A 64-year-old man visited the hospital complaining of lower extremity edema and dyspnea for 4 weeks. Laboratory findings showed no azotemia but hypercholesterolemia, hypoalbuminemia, nephrotic-range proteinuria, and microscopic hematuria. Chest and abdominal computed tomography (CT) revealed no ascites, venous thrombosis, or malignancy with the presence of right-side pleurisy. Biochemical analysis of the pleural fluid was consistent with chylothorax. The patient was confirmed to have MN by percutaneous kidney biopsy. An angiotensin receptor blocker, diuretics, and a hypolipidemic agent were prescribed; non-per os, total parenteral nutrition (TPN), and subcutaneous injection of octreotide were added for management of chylothorax. As serum anti-phospholipase receptor 2 antibody (Ab) concentration increased again, immunosuppressive therapy (IST) consisting of alternating monthly cycles of glucocorticoids and oral cyclophosphamide was instituted. With no improvement in chylothorax and deteriorating nutritional status despite 3 weeks of medical therapy, lymphangiography was performed, followed by thoracic duct embolization (TDE). The patient was discharged from the hospital on day 53 with clinical improvement. At 9 months after discharge, clinical remission of primary MN was achieved without recurrence of chylothorax. CONCLUSIONS: Patients with nephrotic syndrome may rarely exhibit refractory chylothorax without chylous ascites, increasing the risk of serious metabolic complications such as severe malnutrition. Therefore, upon confirming chylothorax associated with primary nephrotic syndrome, prompt radiologic intervention for lymphatic leakage must be considered in addition to specific IST.

Rhabdomyolysis and Acute Kidney Injury Associated with Salmonella Infection: A Report of 2 Cases.

BACKGROUND Rhabdomyolysis is a clinical syndrome characterized by elevated serum creatine kinase (CK) and myoglobin levels due to the breakdown of muscle fibers and is associated with symptoms such as myalgia, muscle swelling, and erythruria. Rhabdomyolysis has an array of potential causes, including Salmonella infection, although rare. We report 2 cases in which nontyphoidal salmonellae caused acute gastroenteritis complicated by rhabdomyolysis and myoglobinuric acute kidney injury (AKI). CASE REPORT Two male patients, aged 69 years and 62 years, presented to our hospital with sudden-onset fever, abdominal pain, and watery diarrhea. At the time of admission, the patients had elevated serum CK levels (32 225 U/L and 10 590 U/L, respectively) and serum creatinine levels (4.8 mg/dL and 8.8 mg/dL, respectively). Both patients also had elevated serum myoglobin concentrations with significant myoglobinuria. They were administered fluid therapy and intravenous empirical antibiotics (cefotaxime and metronidazole for Case 1, ciprofloxacin for Case 2). The patient in Case 2 underwent 3 sessions of hemodialysis due to persistent oliguria and exacerbation of metabolic acidosis. Salmonella B (Case 1) and Salmonella C (Case 2) were isolated from blood cultures. After about 2 weeks of inpatient care, both patients showed improvement of clinical symptoms and were discharged. CONCLUSIONS Patients with acute gastroenteritis induced by Salmonella infection can develop rhabdomyolysis and myoglobinuric AKI in rare cases. Timely administration of appropriate antibiotics and fluids is expected to produce a favorable prognosis. Furthermore, early initiation of hemodialysis after onset of oliguric AKI can improve clinical outcomes.

A forgotten double-J ureteral stent resulting in an emphysematous perinephric abscess: A case report.

RATIONALE: Double-J stents (DJSs) are urologic devices widely used for urinary tract obstruction treatment. Perinephric abscess is a condition with purulent accumulation resulting from urinary tract infection retained between the renal capsule and Gerota's fascia. Emphysematous urinary tract infection in patients with a forgotten DJS is extremely rare. Herein, we report a case of emphysematous perinephric abscess as a complication in a 56-year-old non-diabetic woman who neglected a 10-year-old DJS placed for obstructive uropathy treatment. PATIENT CONCERNS: The patient presented with fever and abdominal pain that persisted for 4 days. Laboratory examinations showed leukocytosis, hypoalbuminemia (2.3 g/dL), and elevated C-reactive protein level (305.5 mg/L) with no azotemia. DIAGNOSIS: Abdominal computed tomography scan revealed a DJS with encrustation and multiple stones in the right kidney as well as a perinephric abscess with gas formation. INTERVENTIONS: Intravenous administration of piperacillin/tazobactam was initiated immediately and percutaneous catheter drainage was performed. Extended-spectrum beta-lactamase-producing Escherichia coli was identified on abscess culture and antibiotics were switched to meropenem, resulting in gradual improvement of the inflammatory lesion. The patient was referred to the urology department for retained DJS removal and vesicolitholapaxy. A piece of fractured stent was removed via open ureterolithotomy. OUTCOMES: Since discharge on hospital day 42, she has been under regular follow-up, and the surgical wound has been healing with no significant sequelae. LESSONS: Prompt medical therapy for inflammation and thorough urologic correction of the stent-induced structural deformities are crucial in long-term neglected DJS and resulting emphysematous perinephric abscess. Patients who undergo DJS placement should be systematically followed up to prevent potential neglect of device management.

Systemic lupus erythematosus presenting as hyponatremia-associated rhabdomyolysis: A case report.

RATIONALE: Systemic lupus erythematosus (SLE) is an autoimmune disease that involves multiple organs and causes various clinical manifestations. Cases of rhabdomyolysis as the initial presentation of SLE are rare, and there are no reported cases of SLE presenting hyponatremia-associated rhabdomyolysis as the first manifestation. Herein, we report a case of SLE with lupus nephritis in a patient with acute hyponatremia-associated rhabdomyolysis. PATIENT CONCERNS: A 44-year-old woman was admitted with complaints of altered consciousness, myalgia, and red-brownish urine that first appeared three days prior. Peripheral blood tests revealed elevated creatine kinase (19,013 IU/L) and myoglobin (5099 U/L) levels and severe hyponatremia (111 mEq/L) with no azotemia. Urinalysis showed nephritic sediments. DIAGNOSIS: Whole-body bone scintigraphy showed increased uptake of radiotracer in the both upper and lower extremities. Serological evaluation revealed the presence of anti-nuclear (speckled pattern, 1:640), anti-double stranded DNA, and anti-Smith antibodies and absence of anti-Jo-1 antibody. A kidney biopsy demonstrated mesangial proliferative (class II) lupus nephritis. INTERVENTIONS: Fluid therapy, including intravenous administration of 3% NaCl, was initiated. After three consecutive days of intravenous methylprednisolone (1 g/d), oral prednisolone (1 mg/kg/d), mycophenolate mofetil, and hydroxychloroquine were administered. OUTCOMES: On day 28, the patient was discharged with marked resolution of SLE-associated symptoms and laboratory findings. Lupus reactivation was not present during the subsequent six-month follow-up. LESSONS: Hyponatremia-associated rhabdomyolysis can be the first manifestation of SLE. Moreover, prompt fluid therapy and timely administration of immunosuppressive agents in SLE patients presenting with hyponatremia and rhabdomyolysis can significantly help alleviate disease activity and improve clinical outcomes.

Anti-glomerular basement membrane disease associated with thin basement membrane nephropathy: A case report.

RATIONALE: Simultaneous occurrence of anti-glomerular basement membrane (anti-GBM) disease and thin basement membrane nephropathy (TBMN), both of which invade the type IV collagen subunits, is very rare. Here, we present the case of a 20-year-old male patient diagnosed with both anti-GBM disease and TBMN upon presenting dyspnea and hemoptysis. PATIENT CONCERNS: No laboratory abnormalities, except arterial hypoxemia (PaO275.4 mmHg) and microscopic hematuria, were present. Chest computed tomography revealed bilateral infiltrations in the lower lung fields; thus, administration of empirical antibiotics was initiated. Gross hemoptysis persisted nonetheless, and bronchoscopy revealed diffuse pulmonary hemorrhage with no endobronchial lesions. Broncho-alveolar lavage excluded bacterial pneumonia, tuberculosis, and fungal infection. DIAGNOSIS: Enzyme-linked immunosorbent assay of his serum was positive for anti-GBM antibody (95.1 U/mL). Human leukocyte antigen (HLA) test was positive for both HLA-DR15/-DR04. Other than diffuse thinning of the GBM (average thickness, 220 nm), index kidney biopsy did not demonstrate any specific abnormalities such as crescent formation. INTERVENTIONS: Methylprednisolone was administered intravenously for 7 consecutive days (500 mg/day), followed by the daily dose of oral prednisolone (80 mg). Cyclophosphamide was also orally administered every day for 3 months (250 mg/day). Following 6 sessions of plasmapheresis, the anti-GBM antibody in serum became negative. OUTCOMES: There was no clinical evidence suggesting recurrence of pulmonary hemorrhage or azotemia during hospitalization and 12-month follow-up period. Twelve months after hospital discharge, oral prednisolone was discontinued. LESSONS: The patients with concurrent anti-GBM disease and TBMN will have a favorable prognosis after proper therapy. However, further research is needed to elucidate the pathogenesis and long-term outcome of the comorbidity of these 2 diseases.

Spontaneous Bacterial Peritonitis in an Adult Patient with Minimal Change Disease.

BACKGROUND Pediatric patients with nephrotic syndrome have a high risk of developing spontaneous bacterial peritonitis (SBP). However, SBP in adults with nephrotic syndrome is very rare. We report a case of SBP induced by Escherichia coli in a 60-year-old male patient on immunosuppressive therapy for the treatment of minimal change disease (MCD). CASE REPORT The patient was hospitalized with abdominal pain and generalized edema that had lasted for 2 weeks. The patient first started treatment with high-dose oral prednisolone after being diagnosed with MCD 6 months ago. Complete remission of nephrotic syndrome was not achieved even after 5 months of treatment. Thus, the treatment was changed to combination therapy with cyclosporine and low-dose prednisolone. At the time of admission, leukocytosis, hypoalbuminemia, decreased serum immunoglobulin G (IgG), azotemia, and nephrotic-range proteinuria were observed. Ascitic fluid analysis showed a leukocyte count of 4960/µL (neutrophils 90%). On the suspicion of SBP associated with MCD, intravenous administration of empirical cefotaxime and supportive therapy were initiated; however, symptoms of peritonitis persisted. Extended-spectrum beta-lactamase-negative E. coli was found in ascites cultures. Laparoscopy-assisted peritoneal biopsy revealed no evidence of fungal infection; however, chronic inflammation without granuloma formation was noted. Afterward, cefotaxime was changed to piperacillin-tazobactam. After 4 weeks of antibacterial therapy, the peritonitis was cured and renal function was improved. CONCLUSIONS Adult patients with steroid-resistant MCD accompanied by refractory ascites, severe hypoalbuminemia, and marked reduction in serum IgG are at a high risk of subsequent SBP and require careful monitoring.

Intestinal tuberculosis in a patient with end-stage renal disease on hemodialysis: A case report.

RATIONALE: Intestinal tuberculosis (TB) is rarely seen in patients with end-stage renal disease (ESRD). We report an intestinal TB case with a clinical presentation similar to that of colon cancer in a patient with ESRD on hemodialysis. PATIENT CONCERNS: A 49-year-old man presented with a 3-month history of general weakness and anorexia. He had been treated for stage 5 chronic kidney disease (CKD) due to diabetic nephropathy for the last 3 years. His blood urea nitrogen and serum creatinine levels were 96.9 and 8.1 mg/dL, respectively, at the time of admission; azotemia was accompanied by severe anemia, hypoalbuminemia, hyperkalemia, and metabolic acidosis. Hemodialysis was initiated for suspected exacerbation of uremia; however, intermittent fever, night sweats, and abdominal discomfort persisted. DIAGNOSES: Abdominal computed tomography (CT) and whole-body F-fluorodeoxyglucose positron emission tomography were indicative of ascending colon cancer with lymph node metastases. However, colonoscopy with biopsy revealed the formation of submucosal caseating granuloma and acid-fast bacillus. INTERVENTIONS: We initiated quadruple therapy with isoniazid, rifampicin, pyrazinamide, and ethambutol. The patient continued the quadruple regimen for the first 2 months before switching to dual therapy and received anti-TB medications for a total of 12 months. OUTCOMES: After 9 months of standard anti-TB chemotherapy, polypoid residual lesions were noted during follow-up colonoscopy. Laparoscopy-assisted ileocecal resection was performed. No findings suggestive of recurrence of colonic TB were observed on follow-up abdominal CT at 6 months after discontinuation of anti-TB medications. LESSONS: If non-specific uremic symptoms persist in patients with advanced CKD, the possibility of extrapulmonary TB such as intestinal TB must be considered. Also, in patients with radiologic suspicion of colon cancer, endoscopy with biopsy should be performed promptly to exclude colonic TB with similar clinical manifestations.

Dapagliflozin-associated euglycemic diabetic ketoacidosis in a patient with type 2 diabetes mellitus: A case report.

RATIONALE: Rare cases of euglycemic diabetic ketoacidosis (eu-DKA) have been reported after the administration of sodium-glucose cotransporter-2 (SGLT-2) inhibitors. No reports have described eu-DKA complicated by hypernatremia due to SGLT-2 inhibitors. PATIENT CONCERNS: A 76-year-old woman with a 40-year history of type 2 diabetes mellitus (DM), for which metformin (1000 mg/day) and dapagliflozin (10 mg/day) were prescribed, presented with malaise, fever, and oliguria. On presentation, her white blood cell count (11,800/µL), serum creatinine (3.2 mg/dL), and C-reactive protein (54 mg/L) were abnormal. Bilateral pyeloureteritis and diffuse paralytic ileus were present. She received intravenous antibiotics and total parenteral nutrition, and was asked to fast. Her renal function and ileus briefly improved. Oral hypoglycemic agents, metformin and dapagliflozin, along with enteral feeding were reinstituted on day 3 of hospitalization. However, on day 6 of hospitalization, the patient developed an altered state of consciousness including confusion, lethargy, and stupor. Several laboratory abnormalities suggestive of ketoacidosis with euglycemia were noted. DIAGNOSES: The patient was diagnosed with eu-DKA accompanied by severe hypernatremia (corrected serum Na concentration, 163 mEq/L) and hypokalemia following dapagliflozin re-administration. INTERVENTIONS: The patient was treated with indicated intravenous fluid therapy. Dapagliflozin use was discontinued. OUTCOMES: The patient's mental status and laboratory findings improved gradually, and she was discharged on maintenance doses of insulin and metformin on day 14 of hospitalization. LESSONS: Acute illnesses such as diffuse paralytic ileus and urinary tract infection, and dietary restrictions or fasting in patients with DM can be considered potential predisposing factors for SGLT-2 inhibitor-associated eu-DKA. For patients with diabetes in the setting of acute morbidity, timely resumption of the SGLT-2 inhibitor therapy should be carefully determined. In addition, eu-DKA due to SGLT-2 inhibitor use may be accompanied by electrolyte disturbances such as hypernatremia and hypokalemia.

Concurrent pituitary apoplexy and posterior reversible encephalopathy syndrome in a patient with end-stage renal disease on hemodialysis: A case report.

RATIONALE: Pituitary apoplexy (PA) and posterior reversible encephalopathy syndrome (PRES) are rare neurologic diseases that show acute neuro-ophthalmologic symptoms such as headache, decreased visual acuity, and altered consciousness. These diseases are rarely found in patients with end-stage renal disease (ESRD) on hemodialysis, and simultaneous occurrence of these 2 diseases has not been reported. PATIENT CONCERNS: The patient was a 75-year-old man with a history of hypertension, diabetes mellitus, and non-functioning pituitary macroadenoma. He had been receiving hemodialysis for ESRD for 3 months before his presentation to the emergency room. The patient complained of headache, vomiting, and dizziness that started after the previous day's hemodialysis. The patient had voluntarily discontinued his antihypertensive medication 2 weeks before presentation and had high blood pressure with marked fluctuation during hemodialysis. Complete ptosis and ophthalmoplegia on the right side suggested 3rd, 4th, and 6th cranial nerve palsies. DIAGNOSES: Magnetic resonance imaging of the brain revealed a pituitary tumor, intratumoral hemorrhage within the sella, and symmetric vasogenic edema in the subcortical white matter in the parieto-occipital lobes. Based on these findings, the patient was diagnosed with PA and PRES. INTERVENTIONS: Intravenous administration of hydrocortisone (50 mg every 6 hours after a bolus administration of 100 mg) was initiated. Although surgical decompression was recommended based on the PA score (5/10), the patient declined surgery. OUTCOMES: Headache and ocular palsy gradually improved after supportive management. The patient was discharged on the 14th day of hospitalization with no recurrence 5 months post-presentation. Current therapy includes antihypertensive agents, oral prednisolone (7.5 mg/day), and maintenance hemodialysis. LESSONS: Neurologic abnormalities developed in a patient with ESRD on hemodialysis, suggesting the importance of prompt diagnosis and treatment in similar instances.

Renal hemosiderosis secondary to intravascular hemolysis after mitral valve repair: A case report.

RATIONALE: Renal hemosiderosis is a disease in which hemosiderin deposits in the renal cortex as a form of iron overload. However, cases of renal hemosiderosis due to intravascular hemolysis following mitral valve repair have been rarely reported. PATIENT CONCERNS: We present the case of a 62-year-old woman who developed asymptomatic urinary abnormalities including microscopic hematuria and proteinuria due to renal hemosiderosis following a mitral valve repair surgery performed two years earlier. DIAGNOSES: A percutaneous renal biopsy showed no specific glomerular abnormality, tubular atrophy, or interstitial fibrosis but extensive deposition of hemosiderin in the proximal tubule. The patient was diagnosed with renal hemosiderosis and chronic intravascular hemolysis following mitral valve repair. INTERVENTIONS: Our patient refused a mitral valve repeat surgery and hence was treated with oral iron preparations, N-acetylcysteine, and a ß-receptor blocker. OUTCOMES: Moderate mitral regurgitation with the regurgitant blood striking against the annuloplasty ring was confirmed on follow-up echocardiography. After the 24-month follow-up period, hemolytic anemia persisted, but there was no significant decline of renal function. LESSONS: For cases of chronic intravascular hemolysis accompanied with asymptomatic urinary abnormalities, a renal biopsy is required to exclude underlying kidney pathology and predict potential renal insufficiency.