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[1例与丙硫氧嘧啶治疗格雷夫斯病相关的髓过氧化物酶-抗中性粒细胞胞浆抗体(ANCA)相关性新月体性肾小球肾炎儿科病例]

[A pediatric case of myeloperoxidase-antineutrophil cytoplasmic (ANCA)-related crescentic glomerulonephritis associated with propylthiouracil treatment for Graves' disease].

作者信息

Kawasaki Y, Suzuki J, Sike T, Isome M, Nozawa R, Suzuki S, Kume K, Suzuki H

机构信息

Department of Pediatrics, Fukushima Medical University School of Medicine, Japan.

出版信息

Nihon Jinzo Gakkai Shi. 1998 Nov;40(8):612-7.

PMID:9893462
Abstract

We treated a 13-year-old girl who developed myeloperoxidase-antineutrophil cytoplasmic antibody (MPO-ANCA)-related crescentic glomerulonephritis (GN) during propylthiouracil (PTU) treatment for Graves' disease. MPO-ANCA-related crescentic GN during PTU therapy has been described previously in only one recent report of 2 children. We report this case here and describe 15 (13 adult cases) more patients with MPO-ANCA-related GN associated with PTU found in a literature review. The mean age at onset was 41.3 years, and the length of PTU administration ranged from 2 weeks to 6 years (mean 3.5 years). Clinical signs and symptoms were hematuria (100%), proteinuria (100%), arthralgia (7 of 16 cases; 43.8%), fever (4 cases; 20.0%), purpura (2 cases; 12.5%), skin ulcer (1 case; 6.3%) and dyspnea (1 case; 6.3%). These patients were treated with steroid (15 cases; 93.8%), cyclophosphamide (8 cases; 50.0%), steroid pulse therapy (4 cases; 25.0%), or plasma exchange (1 case; 6.3%), or were not treated (1 case; 6.3%). Most patients revealed crescentic GN (15 cases; 93.8%) on renal biopsy, while one exhibited mesangial proliferative GN (6.3%). For 2 of the 16 patients (12.5%) irreversible renal dysfunction persisted and hemodialysis was started. Patients with Graves' disease treated with PTU should be observed carefully by urinalysis and monitoring of the serum creatinine level.

摘要

我们治疗了一名13岁女孩,她在接受丙硫氧嘧啶(PTU)治疗格雷夫斯病期间患上了髓过氧化物酶 - 抗中性粒细胞胞浆抗体(MPO - ANCA)相关的新月形肾小球肾炎(GN)。PTU治疗期间的MPO - ANCA相关新月形GN此前仅在最近一篇关于2名儿童的报告中有所描述。我们在此报告该病例,并在文献综述中描述了另外15例(13例成人病例)与PTU相关的MPO - ANCA相关GN患者。发病时的平均年龄为41.3岁,PTU给药时间为2周至6年(平均3.5年)。临床体征和症状包括血尿(100%)、蛋白尿(100%)、关节痛(16例中的7例;43.8%)、发热(4例;20.0%)、紫癜(2例;12.5%)、皮肤溃疡(1例;6.3%)和呼吸困难(1例;6.3%)。这些患者接受了类固醇治疗(15例;93.8%)、环磷酰胺治疗(8例;50.0%)、类固醇脉冲治疗(4例;25.0%)或血浆置换(1例;6.3%),或未接受治疗(1例;6.3%)。大多数患者在肾活检时显示为新月形GN(15例;93.8%),而1例表现为系膜增生性GN(6.3%)。16例患者中有2例(12.5%)持续存在不可逆的肾功能不全并开始进行血液透析。接受PTU治疗的格雷夫斯病患者应通过尿液分析和监测血清肌酐水平进行仔细观察。

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