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Tmem173 N153S/WT 小鼠模型中Ⅰ型干扰素非依赖性 T 细胞损伤与婴儿期起病的 STING 相关血管病(SAVI)。

Type I interferon-independent T cell impairment in a Tmem173 N153S/WT mouse model of STING associated vasculopathy with onset in infancy (SAVI).

机构信息

Department of Pediatrics, University Clinic Carl Gustav Carus, TU Dresden, Dresden, Germany.

Institute of Immunology, Medical Faculty TU Dresden, Dresden, Germany.

出版信息

Clin Immunol. 2020 Jul;216:108466. doi: 10.1016/j.clim.2020.108466. Epub 2020 May 27.

Abstract

STING-associated vasculopathy with onset in infancy (SAVI) is an autoimmune disease caused by heterozygous gain of function mutations of STING (stimulator of interferon genes) that had initially been classified as a type I interferonopathy. We recently reported a genetically engineered mouse strain carrying a common SAVI-associated STING mutation. These STING N153S/WT mice reproduce key features of SAVI, including lung inflammation, loss of T cells in spleen and blood, splenomegaly and thymic hypoplasia. Here we show that αβ T lymphocytopenia is due to disrupted T cell development and is associated with impaired T cell activation and a relative increase in γδ T cell numbers. These alterations were not rescued by additional knockout of the type I IFN receptor (IFNAR1). Collectively, our findings consolidate the concept that constitutive STING signalling leads to a SCID-like phenotype in STING N153S/WT mice.

摘要

婴儿期起病的 STING 相关性血管病(SAVI)是一种由 STING(干扰素基因刺激物)的杂合获得性功能突变引起的自身免疫性疾病,最初被归类为 I 型干扰素病。我们最近报道了一种携带常见 SAVI 相关 STING 突变的基因工程小鼠品系。这些 STING N153S/WT 小鼠再现了 SAVI 的关键特征,包括肺部炎症、脾脏和血液中 T 细胞缺失、脾肿大和胸腺发育不良。在这里,我们表明 αβ T 淋巴细胞减少是由于 T 细胞发育紊乱引起的,与 T 细胞活化受损和 γδ T 细胞数量相对增加有关。这些改变不能通过额外敲除 I 型 IFN 受体(IFNAR1)来挽救。总之,我们的研究结果证实了这样一种概念,即组成性 STING 信号导致 STING N153S/WT 小鼠出现 SCID 样表型。

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