Paediatric Renal Department, NHS Lothian, Edinburgh, UK
Paediatric Renal Department, NHS Lothian, Edinburgh, UK.
BMJ Case Rep. 2022 Apr 20;15(4):e246281. doi: 10.1136/bcr-2021-246281.
A male child presented initially well with a mixed nephrotic-nephritic syndrome and was commenced on standard high-dose oral corticosteroids. Clinical deterioration occurred 3 weeks later with rapidly progressing renal dysfunction, seizures and diminished urinary output, requiring renal replacement therapy. Once stabilised, renal biopsy demonstrated mesangial and capillary C3, minimal IgG deposition, with mesangial electron dense deposits felt consistent with postinfectious glomerulonephritis or C3 glomerulopathy. Further investigations identified circulating autoantibody directed against factor H, as a plausible aetiology of the membranoproliferative glomerulonephritis (MPGN). Treatment with rituximab and mycophenolate mofetil was associated with a reduction in antibody titres and a concurrent reduction in proteinuria and normalisation of renal function.Subsequent monitoring of antibody titres prompted further administrations of rituximab, with reduction in titres demonstrated after repeat doses. Atypical presentations or complications of nephrotic syndrome or MPGN should prompt detailed investigations for the cause with consideration of antifactor H antibodies.
一名男童最初表现为混合性肾病综合征和肾炎综合征,并开始接受标准大剂量口服皮质类固醇治疗。3 周后,临床病情恶化,出现进行性肾功能衰竭、癫痫发作和尿量减少,需要肾脏替代治疗。一旦病情稳定,肾活检显示系膜和毛细血管 C3、最小 IgG 沉积,系膜电子致密沉积物提示与感染后肾小球肾炎或 C3 肾小球病一致。进一步的检查发现,循环自身抗体针对因子 H,可能是膜增生性肾小球肾炎(MPGN)的病因。利妥昔单抗和霉酚酸酯治疗与抗体滴度降低以及蛋白尿减少和肾功能正常化相关。随后监测抗体滴度提示进一步给予利妥昔单抗,重复剂量后滴度降低。肾病综合征或 MPGN 的不典型表现或并发症应提示详细调查病因,并考虑抗因子 H 抗体。