Department of Pathology, Stanford University, H2110, 300 Pasteur Drive, Stanford, CA, 94305, USA.
UC Davis, Sacramento, CA, USA.
Pediatr Nephrol. 2022 Dec;37(12):3127-3137. doi: 10.1007/s00467-022-05503-7. Epub 2022 Mar 25.
Membranous nephropathy is an uncommon cause of nephrotic syndrome in pediatrics.
We reviewed our kidney biopsy records for patients ≤ 20 years of age with membranous nephropathy without evidence of systemic lupus erythematosus within 6 months of biopsy (January 1995-September 2020). Staining for PLA2R, NELL1, THSD7A, SEMA3B, EXT2 (3 biopsies), and IgG-subclass were performed.
Sixteen children (≤ 12 years) and 25 adolescents (13-20 years) were identified. Four children and 15 adolescents showed autoantigen positivity: PLA2R+/SEMA3B- (13), SEMA3B+/PLA2R+ (2), SEMA3B+/PLA2R- (1), NELL1 (1), EXT2+ (2), and THSD7A (0). Co-morbidities associated with PLA2R positivity included IPEX syndrome, active hepatitis B, Von Hippel Lindau syndrome, solitary kidney, type 1 diabetes, hyperuricemia, pregnancy (1), obesity (3), type II diabetes, H. pylori, viral prodrome, and nephrolithiasis. The SEMA3B+/PLA2R- adolescent was pregnant, the NELL1+ adolescent was obese, and the two EXT2+ adolescents eventually met the clinical criteria for lupus (4, 9 years post-biopsy). Co-morbidities among the remaining 24 patients included remote hepatitis B (2), Down's syndrome, lysinuric protein intolerance, recurrent UTIs, hypothyroidism, pregnancy (3), and obesity (2). Follow-up data was available for 12 children and 16 adolescents. Of the 12 children, 6 achieved complete remission, 4 achieved partial remission, and 2 had no response to treatment (1 transplant). Of the 16 adolescents, 4 achieved complete remission, 4 achieved partial remission, and 8 had no response to treatment (3 transplants). A child with "full-house" immunofluorescence staining achieved spontaneous disease remission.
Our non-lupus membranous nephropathy cohort represents one of the largest pediatric studies to date. A higher resolution version of the Graphical abstract is available as Supplementary information.
膜性肾病是儿科肾病综合征的一种罕见病因。
我们回顾了我院在过去 6 个月内(1995 年 1 月至 2020 年 9 月)接受肾活检的、≤ 20 岁且无系统性红斑狼疮证据的膜性肾病患者的活检记录。对 PLA2R、NELL1、THSD7A、SEMA3B、EXT2(3 例活检)和 IgG 亚类进行染色。
共确定了 16 名儿童(≤ 12 岁)和 25 名青少年(13-20 岁)。4 名儿童和 15 名青少年的自身抗原阳性:PLA2R+/SEMA3B-(13)、SEMA3B+/PLA2R+(2)、SEMA3B+/PLA2R-(1)、NELL1(1)、EXT2+(2)和 THSD7A(0)。与 PLA2R 阳性相关的合并症包括 IPEX 综合征、乙型肝炎活动期、VHL 综合征、孤立肾、1 型糖尿病、高尿酸血症、妊娠(1)、肥胖(3)、2 型糖尿病、H. pylori、病毒前驱期和肾结石。SEMA3B+/PLA2R-的青少年处于妊娠状态,NELL1+的青少年肥胖,2 名 EXT2+的青少年最终符合狼疮的临床标准(活检后 4 年和 9 年)。其余 24 名患者的合并症包括乙型肝炎(2)、唐氏综合征、赖氨酸尿蛋白不耐受、复发性尿路感染、甲状腺功能减退、妊娠(3)和肥胖(2)。12 名儿童和 16 名青少年的随访数据可用。12 名儿童中,6 名完全缓解,4 名部分缓解,2 名无反应(1 例移植)。16 名青少年中,4 名完全缓解,4 名部分缓解,8 名无反应(3 例移植)。一名有“满堂红”免疫荧光染色的儿童自发疾病缓解。
我们的非狼疮性膜性肾病队列是迄今为止最大的儿科研究之一。一个高分辨率版本的图表摘要可在补充信息中获得。