Oftedal Bergithe E, Delaleu Nicolas, Dolan David, Meager Anthony, Husebye Eystein S, Wolff Anette S B
Department of Clinical Science, University of Bergen, Bergen, Norway.
KG Jebsen Center for autoimmune diseases, University of Bergen, Bergen, Norway.
FEBS Lett. 2023 May;597(9):1261-1274. doi: 10.1002/1873-3468.14625. Epub 2023 Apr 20.
Autoimmune polyendocrine syndrome type I (APS-1) is caused by mutations in the autoimmune regulator (AIRE) gene and characterised clinically by multiple autoimmune manifestations and serologically by autoantibodies against tissue proteins and cytokines. We here hypothesised that lack of AIRE expression in thymus affects blood immune cells and performed whole-blood microarray analysis (N = 16 APS-I patients vs 16 controls), qPCR verification, and bioinformatic deconvolution of cell subsets. We identified B cell responses as being downregulated in APS-1 patients, which was confirmed by qPCR; these results call for further studies on B cells in this disorder. The type I interferon (IFN-I) pathway was also downregulated in APS-1, and the presence of IFN antibodies is the likely reason for this mild overall downregulation of the IFN-I genes in most APS-1 patients.
自身免疫性多内分泌腺病I型(APS-1)由自身免疫调节因子(AIRE)基因突变引起,临床特征为多种自身免疫表现,血清学特征为针对组织蛋白和细胞因子的自身抗体。我们在此推测,胸腺中AIRE表达的缺失会影响血液免疫细胞,并进行了全血微阵列分析(16例APS-I患者与16例对照)、qPCR验证以及细胞亚群的生物信息解卷积分析。我们发现APS-1患者的B细胞反应下调,这一点通过qPCR得到证实;这些结果需要对该疾病中的B细胞进行进一步研究。I型干扰素(IFN-I)途径在APS-1中也下调,IFN抗体的存在可能是大多数APS-1患者中IFN-I基因总体轻度下调的原因。