Dixit Mehul P, Kirschner Rebecca, Bulimbasic Stella, Dixit Naznin M, Harris Alexis
Florida Children's Kidney Center, University of Central Florida College of Medicine , Orlando, FL , USA.
Nephropath Associates, Little Rock, AR , USA.
NDT Plus. 2010 Oct;3(5):483-6. doi: 10.1093/ndtplus/sfq143. Epub 2010 Jul 27.
Goodpasture's syndrome has been documented in only a handful of children under the age of four. We describe a 3-year-old girl presenting with anaemia and renal failure whose kidney biopsy showed anti-glomerular basement membrane (GBM) disease. She was treated aggressively with pulse steroids, plasmapheresis and monthly infusions of cyclophosphamide. After months of aggressive immunosuppression, her renal function normalized, and her anti-GBM antibody disappeared. A year after the onset, she underwent a second kidney biopsy for persistent proteinuria and hypertension that surprisingly showed focal sclerosing glomerulonephritis, an unreported finding at this age. The biopsy showed deposition of antibody on the GBM despite the fact that anti-GBM antibody had normalized in the serum 5 months earlier. Mycophenolate mofetil was added to the immunosuppression at that point. At her 3-year follow-up, creatinine clearance was 88.4 mL/min/1.73 m(2), proteinuria was 408 mg/day and blood pressure was controlled with enalapril 0.2 mg/kg/day. She has not had a relapse or abnormal anti-GBM antibody for 30 months, but her renal prognosis remains guarded. To our knowledge, this is the youngest patient to have a successful rescue of renal function after isolated Goodpasture's syndrome.
仅在少数4岁以下儿童中记录到了肺出血肾炎综合征。我们描述了一名3岁女孩,她表现为贫血和肾衰竭,肾脏活检显示为抗肾小球基底膜(GBM)病。她接受了大剂量类固醇、血浆置换和每月一次环磷酰胺输注的积极治疗。经过数月的积极免疫抑制治疗后,她的肾功能恢复正常,抗GBM抗体消失。发病一年后,她因持续性蛋白尿和高血压接受了第二次肾脏活检,结果令人惊讶地显示为局灶节段性肾小球硬化,这在该年龄段未见报道。尽管5个月前血清中的抗GBM抗体已恢复正常,但活检仍显示抗体沉积在GBM上。此时在免疫抑制治疗中加用了霉酚酸酯。在她3年的随访中,肌酐清除率为88.4 mL/min/1.73 m²,蛋白尿为408 mg/天,血压通过依那普利0.2 mg/kg/天得到控制。她30个月来未复发,抗GBM抗体也未异常,但她的肾脏预后仍不容乐观。据我们所知,这是孤立性肺出血肾炎综合征后肾功能成功挽救的最年轻患者。