Takeda Yoko, Abe Aya, Toki Takeshi, Komaba Hirotaka, Abe Takaya, Umezu Michio, Joh Kensuke, Fukagawa Masafumi
Division of Nephrology and Kidney Center, Kobe University School of Medicine, Hyogo, Japan.
Nihon Jinzo Gakkai Shi. 2009;51(7):897-903.
In early June 2004, a 50-year-old female was admitted to the hospital for slight fever, general fatigue, hemoptysis, dyspnea, and renal dysfunction (serum creatinine[Cr] : 6.05 mg/dL). She had been treated with prednisolone (PSL : 10-20 mg/day) for RA. She was diagnosed with Goodpasture syndrome based on a high titer of anti-glomerular basement membrane antibody (87 EU), and pulmonary hemorrhage. The renal and pulmonary impairments were markedly improved by the pulse therapy, plasma exchange and temporary hemodialysis. However, the Cr level remained at 2.0 mg/dL, indicating nephrotic syndrome. Light microscopy with Periodic acid-Shiff(PAS) staining demonstrated global sclerosis in three of ten glomeruli. Five glomeruli showed the formation of cellular, and fibrocellular crescents, and the formation of fibrous crescents. Tubular damage and interstitial fibrosis were severe. Immunofluorescence microscopy disclosed major depositions of IgG in a linear pattern along the glomerular basement membrane(GBM). Electron microscopy revealed foot process effacement (>50%)and no electron-dense deposits. Therefore, we diagnosed Goodpasture syndrome associated with minimal change nephrotic syndrome (MCNS). Some reports have dealt with the association of RA and Goodpasture syndrome with D-penicillamine, and of RA and antineutrophil cytoplasmic antibodies (ANCA)-related vasculitis with pulmonary hemorrhage, but none has dealt with cases complicated with RA and Goodpasture syndrome associated with MCNS. Accordingly, whether or not there is a causal relationship between RA and Goodpasture syndrome remains obscure, but since the number of reported cases is small, experience with more cases is necessary to clarify this matter.
2004年6月初,一名50岁女性因低热、全身乏力、咯血、呼吸困难及肾功能不全(血清肌酐[Cr]:6.05mg/dL)入院。她曾因类风湿关节炎接受泼尼松龙治疗(PSL:10 - 20mg/天)。基于高滴度抗肾小球基底膜抗体(87EU)及肺出血,她被诊断为Goodpasture综合征。经脉冲疗法、血浆置换及临时血液透析,肾脏和肺部损害明显改善。然而,Cr水平仍维持在2.0mg/dL,提示肾病综合征。高碘酸 - 希夫(PAS)染色的光镜检查显示,十个肾小球中有三个呈全球硬化。五个肾小球出现细胞性和纤维细胞性新月体形成以及纤维性新月体形成。肾小管损伤和间质纤维化严重。免疫荧光显微镜检查显示IgG沿肾小球基底膜(GBM)呈线性模式大量沉积。电子显微镜检查显示足突消失(>50%)且无电子致密沉积物。因此,我们诊断为与微小病变肾病综合征(MCNS)相关的Goodpasture综合征。一些报告涉及类风湿关节炎与Goodpasture综合征和青霉胺的关联,以及类风湿关节炎与抗中性粒细胞胞浆抗体(ANCA)相关血管炎和肺出血的关联,但均未涉及合并类风湿关节炎和与MCNS相关的Goodpasture综合征的病例。因此,类风湿关节炎与Goodpasture综合征之间是否存在因果关系仍不明确,但由于报告病例数量较少,需要更多病例的经验来阐明此事。