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1/13. nephrotic syndrome associated with Kimura disease.

    Kimura disease presents as benign subcutaneous swelling predominantly around the head and neck region. It has a high incidence of renal involvement. However, the pathogenesis of this association remains elusive. Only 2 pediatric cases and 11 adult cases of Kimura disease with renal involvement have been reported in the literature. In recent years many immunopathogenetic features suggesting an underlying T-cell and related cytokine defect have been noted in Kimura disease. We describe a unique case of an Asian boy who presented with nephrotic syndrome resistant to steroid and cytotoxic therapy, and 5 years later developed cervical lymphadenopathy consistent with Kimura disease. We also review the literature, summarizing the presentation, differential diagnosis, incidence of renal disease, prognosis, immunopathogenetic features, and therapy.
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2/13. Shunt nephritis from propionibacterium acnes in a solitary kidney.

    Since its initial description in 1965, immune complex glomerulonephritis associated with ventriculoatrial shunts (VAS) has been reported widely in the literature. The most common incriminating organism is staphylococcus epidermidis, but less often, an organism generally regarded as nonpathogenic, such as propionibacterium acnes, has been noted as the cause. Shunt infection usually occurs within a few months after placement or manipulation of the shunt, and shunt nephritis (SN) develops gradually over months to years after. Treatment involves mandatory removal of the shunt and antibiotics; prognosis is variable. We report a case of SN with P acnes that is unusual because of its occurrence in a solitary kidney 6 years after shunt placement, persistently negative blood cultures, and normal complement levels. Percutaneous biopsy of a solitary kidney should be considered if it is expected that the result may guide therapy of progressive renal failure.
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3/13. interferon-alpha in combination with ribavirin as initial treatment for hepatitis c virus-associated cryoglobulinemic membranoproliferative glomerulonephritis.

    Mixed cryoglobulinemia (MC) and glomerulonephritis are the most important extrahepatic manifestations of chronic hepatitis c virus (HCV) infection. In HCV-infected patients with MC, renal involvement worsens the overall prognosis because of a high incidence of infection or cardiovascular disease. The relationship between MC and HCV infection has prompted the use of antiviral therapy. Two patients with chronic HCV infection, type-II MC and membranoproliferative glomerulonephritis (MPGN), presenting as nephrotic syndrome were treated with interferon (IFN)-alpha (3 MU 3 times per week) and ribavirin (15 mg/kg daily) for 6 months. Laboratory tests included measurement of anti-HCV antibodies, HCV rna, and HCV genotyping, and characterization of circulating cryoglobulins. A pretreatment renal biopsy was performed, and the histopathologic lesions were scored according to the index of disease activity. viremia and cryoglobulinemia were suppressed in both patients. However, a complete remission of proteinuria was observed in 1 patient only. The evaluation of the renal biopsy specimens revealed a mild MPGN (activity score: 5/24) in the patient with remission of proteinuria and a severe MPGN (activity score: 15/24) in the patient who maintained a nephrotic-range proteinuria. Although a fully satisfactory treatment is not yet available, we feel that a reasonable therapeutic strategy for HCV-infected patients with MC nephritis could be as follows: (1) antiviral treatment alone for patients with a low-grade kidney involvement, and (2) a short-term course of steroids and cytotoxic drugs followed by antiviral therapy for acute exacerbations and/or rapidly progressive GN.
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4/13. Idiopathic myelofibrosis associated with renal extramedullary hematopoiesis and nephrotic syndrome: case report.

    Idiopathic myelofibrosis is characterized by bone marrow fibrosis, anemia, leukoerythroblastosis, and extramedullary hematopoiesis in many organs. Renal abnormalities in idiopathic myelofibrosis have been rarely described in the literature and include extramedullary hematopoiesis in the pararenal or retroperitoneal areas resulting in obstructive uropathy and hemtopoietic cell infiltration in tubulointerstitial area and urolithiasis. These lead to azotemia or acute renal failure, which may respond well to radiotherapy and adjuvant chemotherapy. To our knowledge, there has been only one case report of nephrotic syndrome associated with glomerulonephritis in a myelofibrosis patient; however, no effective treatment was described. Herein, we report the case of a patient with idiopathic myelofibrosis who initially presented with hepatomegaly, anemia, and leukoerythroblastosis. A nephrotic syndrome developed 7 years after initial diagnosis. Renal biopsy disclosed the unique pathological finding of simultaneous mesangial proliferative glomerulonephritis, renal extramedullary hematopoiesis, and gouty nephropathy. Despite treatment with busulfan, proteinuria persisted that implied irreversible glomerular injury and a terminal prognosis. We focus on the unusual pathological finding and the association between nephrotic syndrome and idiopathic myelofibrosis.
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5/13. Membranoproliferative glomerulonephritis.

    glomerulonephritis (GN) encompasses a wide variety of primary and secondary diseases that cause injury to the functioning unit of the kidney, the glomerulus. The many classifications of GN sometimes lead to confusion. This case study describes an individual with membranoproliferative GN and includes discussion of classification, treatment, and prognosis of this disease.
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6/13. Long-term follow-up of non-systemic lupus erythematosus glomerulonephritis in patients with hereditary angioedema: report of four cases.

    Hereditary angioedema (HAE) is characterized by a deficiency in C1 inhibitor protein (C1 INH) and by clinical symptoms of episodic swelling of subcutaneous or mucosal tissue. It has rarely been reported in association with non-systemic lupus erythematosus (SLE) glomerulonephritis (GN). A recent report of two cases indicates the prognosis to be poor, with both patients progressing to chronic renal failure 8 and 20 years after diagnosis. This report describes the 5-year follow-up of a previously unreported case of an 8-year-old boy with HAE and non-SLE membranoproliferative glomerulonephritis (MPGN). The patient developed macroscopic hematuria, azotemia, and a vasculitic rash. Treatment included prednisone and cyclophosphamide, resulting in clinical improvement. The present report also summarizes the long-term follow-up of three previously reported cases of HAE and non-SLE GN, 25, 16, and 10 years after their initial presentation. patients monitored for 25 and 16 years had MPGN and normal renal function and received no therapy. The third patient, monitored for 10 years, had segmental MPGN. This patient presented with urinary abnormalities and, after treatment with prednisone, had improvement in her hematuria. None of these four patients developed chronic renal failure. These observations indicate a variable outcome in patients with HAE and non-SLE GN.
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7/13. Crescentic glomerulonephritis associated with bacterial endocarditis--antibiotics alone may be sufficient. A case report.

    Crescentic glomerulonephritis complicating the course of bacterial endocarditis carries a poor prognosis. Ideal treatment strategy is not clearly defined. In addition to antibiotic treatment, plasmapheresis and steroids have been used with variable results. Here we report a case of 40-year old female who was referred because of generalized body swelling and decrease urine output associated with low grade fever on and off for two to three months. She was diagnosed to have acute renal failure secondary to tricuspid valve endocarditis. Staph aureus was isolated from blood culture and renal biopsy showed crescentic glomerulonephritis. She received dialysis support and antibiotics and had complete recovery of renal function 6 weeks after initiation of therapy. Eradication of infection with antibiotics treatment may be sufficient for resolution of crescentic glomerulonephritis associated with infective endocarditis in some cases.
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8/13. Sequential observation of fundus changes in patients with long standing membranoproliferative glomerulonephritis type II (MPGN type II).

    Specific fundus changes have been reported in patients with membranoproliferative glomerulonephritis type II (MPGN type II). We studied the clinical course of this retinopathy in four patients who all had a long follow-up with several fundus examinations. Sequential observation was indicative of a slow progression of the retinopathy. Most eyes maintained in the chronic stages a nearly normal visual acuity, and a full visual field despite the existence of marked drusen and atrophic changes. The prognosis however must be somewhat guarded, since choroidal neovascularization developed in three eyes and caused bilateral severe visual loss in one patient.
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9/13. Severe hypertension after liver transplantation in alpha 1 antitrypsin deficiency.

    Five children with alpha 1 antitrypsin deficiency and terminal liver disease received liver grafts; all five became hypertensive and four developed hypertensive encephalopathy. There was evidence of renal disease preoperatively and renal biopsy specimens showed variable glomerulonephritic histology with IgA nephropathy in one, mesangial-proliferative changes in two, and mesangio-capillary glomerulonephritis type I in two. Four hypertensive episodes were preceded by a fall in creatinine clearance. The association of glomerulonephritis with alpha 1 antitrypsin deficiency in children is more common than has been recognised. Affected patients are prone to severe hypertension of probable renal origin after liver transplantation and the renal lesion may affect long term prognosis.
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10/13. Idiopathic polymyositis complicated by arthritis and mesangial proliferative glomerulonephritis: case report and review of the literature.

    A 56-yr-old male developed a systemic illness while receiving cyclical oral etidronate therapy for idiopathic osteoporosis. The illness, characterized by fever, proximal myopathy and inflammatory synovitis, was associated with interstitial lung disease and mesangial proliferative glomerulonephritis. Elevated plasma creatine phosphokinase level and inflammatory muscle biopsy findings confirmed a diagnosis of polymyositis (PM). antibodies to Jo-1 were also detected. A review of the literature reveals that mesangial proliferation is the commonest glomerular lesion and suggests a possible association between arthritis and glomerulonephritis in PM. The prognosis of this renal lesion appears to be good, although only limited data is available.
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