Department of Rheumatology and Immunology, Ruijin Hospital, Shanghai Jiao Tong University School of Medicine, Shanghai, 200025, China.
Department of Rheumatology, Shanghai Guanghua Hospital of Integrative Medicine, Shanghai, 200052, China; Institute of Arthritis Research in Integrative Medicine, Shanghai Academy of Traditional Chinese Medicine, Shanghai, 201203, China.
J Autoimmun. 2022 Feb;127:102793. doi: 10.1016/j.jaut.2022.102793. Epub 2022 Feb 2.
Adult-onset Still's disease (AOSD) is a rare but clinically well-known auto-inflammatory disorder. Cytokine storm, the hallmark of AOSD, is mediated by neutrophil hyperactivation and enhanced neutrophil extracellular trap (NET) formation. Type I interferons (IFNs), having a primary role in the initiation of proinflammation responses, can induce subsequent inflammatory cytokine production. However, the role of type I IFNs in AOSD is unclear. Indeed, high levels of IFN-α and IFN-β expression are presented by AOSD patients. In this investigation, hierarchical unsupervised clustering was performed on IFN-α and IFN-β data to identify a cluster of AOSD patients who had a serious condition. Neutrophils from treatment-naïve active AOSD patients showed very strong enrichment in their IFN-α response, as shown by RNA-seq and confirmed by the IFN score. Whether IFN-α stimulates NET formation was also tested. IFN-α had the ability to form NETs that contained oxidized mitochondrial DNA (ox-mtDNA). Moreover, the JAK inhibitor could be used to dampen type I IFN-induced NET formation and eventually control ox-mtDNA release. Our results demonstrated the important roles of type I IFNs in the pathogenesis of AOSD through their promotion of NET formation, as characterized by the enhanced level of ox-mtDNA. The findings open up new avenues of research into therapeutic approaches for AOSD.
成人Still 病(AOSD)是一种罕见但临床上众所周知的自身炎症性疾病。细胞因子风暴是 AOSD 的标志,由中性粒细胞过度激活和增强的中性粒细胞胞外陷阱(NET)形成介导。I 型干扰素(IFN)在炎症反应的启动中起主要作用,可诱导随后的炎症细胞因子产生。然而,I 型 IFNs 在 AOSD 中的作用尚不清楚。事实上,AOSD 患者表现出高水平的 IFN-α 和 IFN-β 表达。在这项研究中,对 IFN-α 和 IFN-β 数据进行了分层无监督聚类,以识别具有严重病情的 AOSD 患者聚类。来自未经治疗的活动性 AOSD 患者的中性粒细胞在 IFN-α 反应中表现出很强的富集,这通过 RNA-seq 显示,并通过 IFN 评分得到证实。IFN-α 是否刺激 NET 形成也进行了测试。IFN-α 具有形成 NET 的能力,其中包含氧化的线粒体 DNA(ox-mtDNA)。此外,JAK 抑制剂可用于抑制 I 型 IFN 诱导的 NET 形成,并最终控制 ox-mtDNA 释放。我们的研究结果表明,I 型 IFNs 通过促进 NET 形成在 AOSD 的发病机制中起重要作用,其特征是 ox-mtDNA 水平增强。这些发现为 AOSD 的治疗方法开辟了新的研究途径。