Department of Rheumatology and Immunology, Hannover Medical University, Hannover, Germany; Hannover Medical School, Cluster of Excellence RESIST (EXC 2155), Hannover, Germany.
Department of Rheumatology and Immunology, Hannover Medical University, Hannover, Germany; Hannover Medical School, Cluster of Excellence RESIST (EXC 2155), Hannover, Germany.
EBioMedicine. 2023 Apr;90:104501. doi: 10.1016/j.ebiom.2023.104501. Epub 2023 Mar 2.
Inborn errors of immunity (IEIs) are immunological disorders characterized by variable susceptibility to infections, immune dysregulation and/or malignancies, as a consequence of damaging germline variants in single genes. Though initially identified among patients with unusual, severe or recurrent infections, non-infectious manifestations and especially immune dysregulation in the form of autoimmunity or autoinflammation can be the first or dominant phenotypic aspect of IEIs. An increasing number of IEIs causing autoimmunity or autoinflammation, including rheumatic disease have been reported over the last decade. Despite their rarity, identification of those disorders provided insight into the pathomechanisms of immune dysregulation, which may be relevant for understanding the pathogenesis of systemic rheumatic disorders. In this review, we present novel IEIs primarily causing autoimmunity or autoinflammation along with their pathogenic mechanisms. In addition, we explore the likely pathophysiological and clinical relevance of IEIs in systemic rheumatic disorders.
先天性免疫缺陷(IEI)是一种免疫性疾病,其特征是由于单基因中的种系变异导致对感染、免疫失调和/或恶性肿瘤的易感性不同。虽然最初是在具有异常、严重或复发性感染的患者中发现的,但非传染性表现,特别是自身免疫或自身炎症形式的免疫失调可能是 IEI 的第一个或主要表型方面。在过去十年中,报道了越来越多的自身免疫或自身炎症的 IEI,包括风湿性疾病。尽管它们很罕见,但这些疾病的鉴定为免疫失调的发病机制提供了深入了解,这可能与理解系统性风湿性疾病的发病机制有关。在这篇综述中,我们主要介绍了主要引起自身免疫或自身炎症的新型 IEI 及其发病机制。此外,我们还探讨了 IEI 在系统性风湿性疾病中的可能病理生理学和临床相关性。