Literature DB >> 30872080

cGAS activation causes lupus-like autoimmune disorders in a TREX1 mutant mouse model.

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.
Copyright © 2019 Elsevier Ltd. All rights reserved.

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


  22 in total

1.  TREX1 - Apex predator of cytosolic DNA metabolism.

Authors:  Sean R Simpson; Wayne O Hemphill; Teesha Hudson; Fred W Perrino
Journal:  DNA Repair (Amst)       Date:  2020-06-12

Review 2.  Intracellular Sensing of DNA in Autoinflammation and Autoimmunity.

Authors:  Susan MacLauchlan; Katherine A Fitzgerald; Ellen M Gravallese
Journal:  Arthritis Rheumatol       Date:  2022-09-06       Impact factor: 15.483

3.  Structural basis for nucleosome-mediated inhibition of cGAS activity.

Authors:  Duanfang Cao; Xiaonan Han; Xiaoyi Fan; Rui-Ming Xu; Xinzheng Zhang
Journal:  Cell Res       Date:  2020-10-13       Impact factor: 25.617

4.  T Cells Produce IFN-α in the TREX1 D18N Model of Lupus-like Autoimmunity.

Authors:  Sean R Simpson; Stephen L Rego; Scott E Harvey; Mingyong Liu; Wayne O Hemphill; Rajkumar Venkatadri; Rahul Sharma; Jason M Grayson; Fred W Perrino
Journal:  J Immunol       Date:  2019-12-11       Impact factor: 5.422

Review 5.  Chemical and Biomolecular Strategies for STING Pathway Activation in Cancer Immunotherapy.

Authors:  Kyle M Garland; Taylor L Sheehy; John T Wilson
Journal:  Chem Rev       Date:  2022-02-02       Impact factor: 60.622

Review 6.  Regulation and inhibition of the DNA sensor cGAS.

Authors:  Jonny Hertzog; Jan Rehwinkel
Journal:  EMBO Rep       Date:  2020-11-05       Impact factor: 9.071

7.  Identification of key biomarkers and immune infiltration in systemic lupus erythematosus by integrated bioinformatics analysis.

Authors:  Xingwang Zhao; Longlong Zhang; Juan Wang; Min Zhang; Zhiqiang Song; Bing Ni; Yi You
Journal:  J Transl Med       Date:  2021-01-19       Impact factor: 5.531

Review 8.  The Trinity of cGAS, TLR9, and ALRs Guardians of the Cellular Galaxy Against Host-Derived Self-DNA.

Authors:  Vijay Kumar
Journal:  Front Immunol       Date:  2021-02-11       Impact factor: 7.561

9.  TREX1 as a Novel Immunotherapeutic Target.

Authors:  Wayne O Hemphill; Sean R Simpson; Mingyong Liu; Freddie R Salsbury; Thomas Hollis; Jason M Grayson; Fred W Perrino
Journal:  Front Immunol       Date:  2021-04-01       Impact factor: 7.561

10.  Small molecule inhibition of human cGAS reduces total cGAMP output and cytokine expression in cells.

Authors:  Caroline Wiser; Byungil Kim; Jessica Vincent; Manuel Ascano
Journal:  Sci Rep       Date:  2020-05-05       Impact factor: 4.379

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