Oleson Ileisa, Fecker Adeline, Richardson Kelsey, Bauer Abbie, Andeen Nicole K, Kung Vanderlene L
School of Medicine, Oregon Health & Science University, Portland, OR, USA.
Division of Pediatric Nephrology and Hypertension, Oregon Health & Science University, Portland, OR, USA.
Pediatr Nephrol. 2024 Mar;39(3):741-744. doi: 10.1007/s00467-023-06172-w. Epub 2023 Oct 19.
Antineutrophil cytoplasmic antibody (ANCA)-associated vasculitis (AAV) and IgG4-related disease (IgG4-RD) are distinct immune disorders with overlapping clinical and laboratory features. While ANCA positivity excludes IgG4-RD in the 2019 ACR/EULAR classification, this criterion is not uniformly applied, and AAV can form inflammatory masses in various organs and show increase in IgG4 + plasma cells, similar to IgG4-RD.
CASE DIAGNOSIS/TREATMENT: A 5-year-old female with history of orbital mass diagnosed as IgG4-RD presents with acute kidney injury. She has a myeloperoxidase ANCA, and kidney biopsy shows pauci-immune crescentic glomerulonephritis and acute tubulointerstitial nephritis with increased IgG4 + plasma cells and tubular basement membrane (TBM) deposits.
In isolation, TBM deposits and increased IgG4 + plasma cells are suggestive of IgG4-RD. In the context of a positive ANCA and pauci-immune crescentic glomerulonephritis, however, increased IgG4 + plasma cells due to AAV are favored. In cases with features of IgG4-RD, ANCA positivity suggests an alternate diagnosis of AAV to be more likely.
抗中性粒细胞胞浆抗体(ANCA)相关性血管炎(AAV)和IgG4相关性疾病(IgG4-RD)是具有重叠临床和实验室特征的不同免疫性疾病。虽然在2019年美国风湿病学会/欧洲抗风湿病联盟(ACR/EULAR)分类中ANCA阳性可排除IgG4-RD,但该标准并未得到统一应用,并且AAV可在各个器官形成炎性肿块,并显示IgG4 +浆细胞增多,类似于IgG4-RD。
病例诊断/治疗:一名有眼眶肿块病史、被诊断为IgG4-RD的5岁女性出现急性肾损伤。她存在髓过氧化物酶ANCA,肾脏活检显示寡免疫性新月体性肾小球肾炎和急性肾小管间质性肾炎,伴有IgG4 +浆细胞增多和肾小管基底膜(TBM)沉积。
单独来看,TBM沉积和IgG4 +浆细胞增多提示IgG4-RD。然而,在ANCA阳性和寡免疫性新月体性肾小球肾炎的背景下,更倾向于AAV导致的IgG4 +浆细胞增多。在具有IgG4-RD特征的病例中,ANCA阳性表明更可能的诊断是AAV。