| Literature DB >> 30872080 |
Nanyang Xiao1, Jingjing Wei1, Shan Xu1, Hekang Du1, Miaohui Huang1, Sitong Zhang1, Weiwei Ye1, Lijun Sun2, Qi Chen3.
Abstract
TREX1 encodes a major cellular DNA exonuclease. Mutations of this gene in human cause cellular accumulation of DNA that triggers autoimmune diseases including Aicardi-Goutieres Syndrome (AGS) and systemic lupus erythematosus (SLE). We created a lupus mouse model by engineering a D18 N mutation in the Trex1 gene which inactivates the enzyme and has been found in human patients with lupus-like disorders. The Trex1D18N/D18N mice exhibited systemic inflammation that consistently recapitulates many characteristics of human AGS and SLE. Importantly, ablation of cGas gene in the Trex1D18N/D18N mice rescued the lethality and all detectable pathological phenotypes, including multi-organ inflammation, interferon stimulated gene induction, autoantibody production and aberrant T-cell activation. These results indicate that cGAS is a key mediator in the autoimmune disease associated with defective TREX1 function, providing additional insights into disease pathogenesis and guidance to the development of therapeutics for human systemic autoimmune disorders.Entities:
Keywords: Autoimmune disorder; DNA sensor; IFN-signature; Innate immunity; Lupus; Trex1 mutation; cGAS
Year: 2019 PMID: 30872080 DOI: 10.1016/j.jaut.2019.03.001
Source DB: PubMed Journal: J Autoimmun ISSN: 0896-8411 Impact factor: 7.094