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学科主题: 临床医学
题名:
Coexistence of Anti-Glomerular Basement Membrane Antibodies and Anti-Neutrophil Cytoplasmic Antibodies in a Child With Human Leukocyte Antigen Susceptibility and Detailed Antibody Description A Case Report
作者: Xie, Li-jun1,2,3,4,6; Cui, Zhao1,2,3,4,5; Jia, Xiao-yu1,2,3,4; Chen, Zhi; Liu, Xiao-rong5; Zhao, Ming-hui1,2,3,4,7
刊名: MEDICINE
发表日期: 2015-07-01
DOI: 10.1097/MD.0000000000001179
卷: 94, 期:29
收录类别: SCI
文章类型: Article
WOS标题词: Science & Technology
类目[WOS]: Medicine, General & Internal
研究领域[WOS]: General & Internal Medicine
关键词[WOS]: PULMONARY RENAL SYNDROME ; SEQUENTIAL DEVELOPMENT ; GBM ANTIBODIES ; DISEASE ; GLOMERULONEPHRITIS ; AUTOANTIBODIES ; ANCA ; MYELOPEROXIDASE ; VASCULITIS ; SPECIFICITY
英文摘要:

Anti-glomerular basement membrane (anti-GBM) disease and anti-neutrophil cytoplasmic antibody (ANCA)-associated vasculitis both could cause rapidly progressive glomerulonephritis. The coexistence of ANCAs and anti-GBM antibodies was known as double positive, which was extremely rare in children. We report a pediatric case with coexistence of ANCAs and anti-GBM antibodies.A 6-year-old girl presented with acute renal failure, hematuria, proteinuria, and oliguria. She was double positive of ANCAs specific to myeloperoxidase, and anti-GBM antibodies. Kidney biopsy confirmed linear immunoglobulin (Ig)G deposit along GBM and 100% of crescent formation in glomeruli; among them 83.3% were cellular crescents. Human leukocyte antigen (HLA) gene typing showed DRB11501, an allele strongly associated with anti-GBM disease, and DRB10405, an independent risk factor for renal failure in patients with ANCA-associated vasculitis. The titer of anti-GBM antibodies was 1:800, and the predominant IgG subclass was IgG1, which was closely related with severe kidney injury and worse outcome. The target antigen of anti-GBM antibodies was restricted on the noncollagen domain 1 of the 3 chain of type IV collagen (3[IV]NC1), with recognitions to both epitopes, E-A (3(17-31)) and E-B (3(127-141)).This is the first reported pediatric case with coexistence of ANCAs and anti-GBM antibodies, in which the HLA typing and immunologic characters of autoantibodies were identified. The findings on this early-onset patient are meaningful for understanding the mechanisms of both anti-GBM disease and ANCA-associated vasculitis.

语种: 英语
所属项目编号: 2012CB517702 ; 81321064 ; 81170645 ; 81330020 ; 81370801 ; 81400703 ; Z121107001012052
项目资助者: Chinese 973 Program ; Natural Science Fund of China ; National Natural Science Fund of China ; Beijing Municipal Science and Technology Commission
WOS记录号: WOS:000359921500001
Citation statistics:
内容类型: 期刊论文
URI标识: http://ir.bjmu.edu.cn/handle/400002259/59176
Appears in Collections:北京大学第一临床医学院_肾脏内科_期刊论文

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作者单位: 1.Peking Univ, Inst Nephrol, Beijing, Peoples R China
2.Minist Hlth China, Key Lab Renal Dis, Beijing, Peoples R China
3.Peking Univ, Hosp 1, Dept Med, Div Renal, Beijing, Peoples R China
4.Minist Hlth China, Key Lab CKD Prevent & Treatment, Beijing, Peoples R China
5.Capital Med Univ, Beijing Childrens Hosp, Dept Nephrol, Beijing 100045, Peoples R China
6.Guangxi Med Univ, Affiliated Hosp 1, Dept Med, Div Renal, Nanning 530021, Guangxi Zhuang, Peoples R China
7.Peking Tsinghua Ctr Life Sci, Beijing, Peoples R China

Recommended Citation:
Xie, Li-jun,Cui, Zhao,Jia, Xiao-yu,et al. Coexistence of Anti-Glomerular Basement Membrane Antibodies and Anti-Neutrophil Cytoplasmic Antibodies in a Child With Human Leukocyte Antigen Susceptibility and Detailed Antibody Description A Case Report[J]. MEDICINE,2015,94(29).
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