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Ai Takahashi,
Yukihiko Kawasaki,
Kazuo Yoshida,
Kazuhiro Mochizuki,
Masato Isome,
Ken Honzumi,
Ruriko Nozawa,
Shigeo Suzuki,
Mitsuaki Hosoya,
Junzo Suzuki,
Hitoshi Suzuki
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ABSTRACT: Viruses have been suspected to be one of the causes of IgA nephropathy (IgAN). Recent studies have detected viruses in renal tissues of patients with IgAN. Enteroviruses have been reported as pathogenic agents in some renal diseases. We previously reported that group B coxsackieviruses cause pathological changes in experimentally infected mouse kidney. The aim of the present study was to examine the participation of enteroviruses in the pathogenesis of renal diseases including IgAN. Renal biopsies of ten patients with IgAN (group 1) and of 19 patients with non-IgAN renal disease (group 2) were analyzed by polymerase chain reaction (PCR) for the presence of enteroviral RNA. Positive PCR results were obtained for three patients (30%) of group 1. We confirmed by sequencing that the positive PCR products were derived from strains of enteroviruses. One of these three patients also had a positive result for lymphocytes from peripheral blood. In contrast, enteroviral RNA was detected in none of the 19 patients of group 2. The incidence of enteroviral RNA detection in patients of group 1 was higher than that in group 2 (P<0.05). Our findings suggest that enteroviral infection may have the possibility of becoming one of the factors involved in the mechanism of onset or evolution of IgAN.
Pediatric Nephrology 11/2005; 20(11):1578-82. · 2.52 Impact Factor
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ABSTRACT: We investigated the efficacy of oral mizoribine pulse therapy (mizoribine-pulse) for cyclosporin (CyA)-dependent, steroid-resistant nephrotic syndrome (SRNS) and frequently relapsing, steroid-dependent nephrotic syndrome (FR-SDNS).
One child with CyA-dependent SRNS and eight children with CyA-dependent FR-SDNS were treated with mizoribine-pulse (daily dose: 10 mg/kg; maximum total dose 500 mg). We compared clinical manifestations before and after mizoribine-pulse, and studied the changes in serum mizoribine concentration in each patient on days when mizoribine was administered.
Four patients had no subsequent relapses (responders). Two of the four responders discontinued prednisolone and CyA, the other two discontinued CyA. Although each of the five other patients (non-responders) experienced single subsequent relapses, the dosages of prednisolone and CyA after mizoribine-pulse were decreased significantly compared with before mizoribine-pulse. The peak blood concentration of mizoribine in the responders was higher than in the non-responders (3.6+/-0.9 vs 1.8+/-0.4 microg/ml).
Mizoribine-pulse may be effective for some patients with CyA-dependent SRNS and FR-SDNS.
Nephrology Dialysis Transplantation 11/2005; 20(10):2243-7. · 3.40 Impact Factor
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ABSTRACT: Selective IgA deficiency associated with glomerulonephritis is rare and no previous reports in childhood have been made of the association of IgA deficiency and membranous glomerulonephritis (MGN). We report a 5-year-old boy with selective IgA deficiency and MGN. He presented with nephrotic syndrome. Percutaneous renal needle biopsy showed diffuse global thickening on light microscopy and heavy IgG and moderate C3 deposits were found on immunofluorescence. Electron microscopy detected extensive global subepithelial deposition of electron-dense material with frequent intramembranous extension and spike formation. The pathological diagnosis was diffuse MGN stage 1. Oral prednisolone (1 mg kg(-1) day(-1)), angiotensin-converting enzyme inhibitors (ACEI), and angiotensin II receptor blocker (ARB) were given resulting in reduction of proteinuria. The prednisolone dose was gradually tapered and discontinued after 2 months. At present the patient has been in complete remission for 10 months despite the discontinuance of prednisolone. In conclusion, our treatment with corticosteroid, ACEI and ARB reduced proteinuria and was effective for our case with selective IgA deficiency and MGN.
Pediatric Nephrology 06/2005; 20(5):662-4. · 2.52 Impact Factor
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ABSTRACT: To clarify the role of subclasses of macrophages in chronic glomerulonephritis, we evaluated the relationship between myeloid-related protein 8 (MRP8) and MRP14 expression on macrophages and the progression of membranoproliferative glomerulonephritis (MPGN).
We enrolled 35 patients with MPGN type 1 who had a normal creatinine clearance at the time of their first renal biopsy and divided them into 2 groups based on clinical status at the time of their most recent examination: 12 patients with normal urine test results and 12 patients with minor urinary abnormalities at the latest observation (group 1) and 7 patients with persistent nephropathy and 4 patients with renal insufficiency (group 2). The first and second renal biopsy findings and MRP8 and MRP14 expression on macrophages were investigated in both groups.
Mean scores for positive glomerular and interstitial MRP8 and CD68 staining at the time of the first and second biopsies were significantly higher in group 2 than group 1. At the time of the second biopsy, mean scores for interstitial CD68-positive (CD68 +) staining were higher in group 2 than group 1. Mean scores for glomerular and interstitial MRP8 + and CD68 + staining at the time of the first biopsy correlated with the chronicity index at the time of second biopsy in both groups.
Results suggest that MRP8 expression on macrophages in glomeruli and interstitial lesions at the first biopsy can be a useful prognostic marker for renal dysfunction in children with MPGN type 1.
American Journal of Kidney Diseases 04/2005; 45(3):510-8. · 5.43 Impact Factor
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ABSTRACT: Hemophagocytic syndrome (HPS) is an unusual but severe illness associated with a variety of infections, as well as genetic, malignant tumors, and autoimmune diseases. We report an 11-year-old girl with systemic lupus erythematosus and nephritis who developed HPS associated with Epstein-Barr virus reactivation. In our patient, the onset of reactive HPS might be related to immunosuppressive treatment during the course of lupus nephritis.
Pediatric Nephrology 03/2005; 20(2):226-8. · 2.52 Impact Factor
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ABSTRACT: There have been reports of the use of mizoribine (MZB) oral pulse therapy for the treatment of systemic lupus erythematosus. We report its efficacy in a 9-year-old girl with steroid- and cyclosporine-dependent nephrotic syndrome (NS). The patient experienced relapses of NS when prednisolone was tapered to 20 mg/day after discontinuing cyclosporine due to biopsy proven toxicity. When methylprednisolone pulse therapy combined with prednisolone therapy (40 mg/day) failed to result in a complete remission after 3 weeks, oral MZB pulse therapy (total dose of 500 mg, 10 mg/kg per day in three divided daily doses twice a week) was given. This therapy was continued for 9 months and resulted in complete remission of the NS for 6 months despite the discontinuation of prednisolone. The serum concentration of MZB was above 2.5 microg/ml for about 10 h (from 3 h after the first dose of MZB to 2 h after the final dose). Thus, our results suggest that this regimen may be effective for patients with steroid-dependent NS.
Pediatric Nephrology 02/2005; 20(1):96-8. · 2.52 Impact Factor
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ABSTRACT: Six Japanese children with rapidly progressive Henoch-Schönlein purpura nephritis (HSPN) received multiple drug therapy combined with plasmapheresis (PP). After five courses of PP, multiple drug therapy, including methylprednisolone and urokinase pulse therapy, oral prednisolone, cyclophophamide, dipyridamole, and warfarin was given. At presentation, urine protein excretion and histological indices of the mean activity and chronicity were 245+/-101 mg/m(2) per hour, 6.6+/-1.2, and 1.5+/-1.3, respectively. After 6 months of therapy, urinary protein excretion had decreased significantly ( P<0.001). The activity index decreased significantly at the second renal biopsy performed at a mean interval of 4.3 months after the first (2.8+/-1.4, P<0.05), while the chronicity index did not change. At the most recent observation, all showed clinical improvement. Two patients had normal urine, three had proteinuria of <20 mg/m(2) per hour, one had proteinuria of >20 mg/m(2) per hour, and none had renal insufficiency. Although this case series is without controls, our treatment protocol may be of benefit to children with rapidly progressive HSPN.
Pediatric Nephrology 08/2004; 19(8):920-3. · 2.52 Impact Factor
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ABSTRACT: We present a case report of a 10 years old boy with protein-losing enteropathy and eosinophilic gastroenteritis who had positive histamine release tests, increased allergen-specific IgE antibodies to some food items, and low levels of total serum protein and albumin. Upper gastrointestinal endoscopy revealed a number of polyps and diffuse gastritis. Biopsy specimens of the stomach and duodenum showed widespread eosinophilia and neutrophilia. Although a restricted diet was recommended, a diet which excluded foods with positive results to both histamine release test and allergen-specific IgE antibodies was poorly tolerated, and the patient rejected systemic administration of corticosteroids. Thus, we initiated an oral disodium cromoglycate (DSCG) and ketotifen therapy. After oral DSCG and ketotifen administration, the patient's condition improved gradually. Therefore, oral DSCG and ketotifen therapy might be considered as treatment option in patients with eosinophilic gastroenteritis and protein-losing enteropathy caused by food allergy.
Asian Pacific journal of allergy and immunology / launched by the Allergy and Immunology Society of Thailand 10/2003; 21(3):193-7. · 0.65 Impact Factor
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ABSTRACT: To evaluate the efficacy of prednisolone, warfarin, and dipyridamole therapy combined with mizoribine (PWDM) in the treatment of diffuse immunoglobulin A (IgA) nephropathy in comparison with prednisolone, warfarin, and dipyridamole therapy without mizoribine (PWD) and with methylprednisolone pulse therapy (PWD pulse).
We collected data on 61 patients diagnosed with diffuse IgA nephropathy, and these patients were retrospectively divided into three groups without randomization. Group A included 21 patients before 1987 who were treated with PWD for 24 months, group B included 20 patients from 1987 to 1989 who were treated with PWD pulse therapy for 24 months, and group C included 20 patients after 1990 who were treated with PWDM for 24 months. Clinical features and pathological findings in each group were analyzed retrospectively.
The time from initiation of therapy in group A, group B, and group C was 8.9 +/- 5.2, 8.1 +/- 3.9, and 7.7 +/- 3.8 years, respectively. At the latest follow-up examination, the mean urinary protein excretion (mg/m2/h) was 17 +/- 10 in group A, 22 +/- 20 in group B, and 6 +/- 6 in group C and had decreased significantly in group C as compared with the other groups. The activity index in all three groups was lower at the second biopsy than that at the first biopsy (5.1 +/- 0.8 vs. 6.5 +/- 2.1 in group A, p < 0.05; 5.6 +/- 0.9 vs. 6.6 +/- 1.7 in group B, p < 0.01, and 4.5 +/- 1.0 vs. 6.8 +/- 1.9 in group C, p < 0.01). The chronicity index in groups A and B at second biopsy was higher than at first biopsy (7.3 +/- 1.4 vs. 4.8 +/- 1.0 in group A, p < 0.01, and 8.1 +/- 2.0 vs. 5.3 +/- 0.9 in group B, p < 0.01), but was unchanged in group C. At the latest follow-up examination, 1 patient (4.8%) in group A, 3 patients (15%) in group B, and none (0%) in group C had renal insufficiency.
These results suggest that PWDM appears to be more effective than PWD or PWD pulse in ameliorating proteinuria and histological severity of patients with IgA nephropathy.
American Journal of Nephrology 24(6):576-81. · 2.54 Impact Factor
