Adenosine-to-inosine editing of endogenous Z-form RNA by the deaminase ADAR1 prevents spontaneous MAVS-dependent type I interferon responses

Immunity. 2021 Sep 14;54(9):1961-1975.e5. doi: 10.1016/j.immuni.2021.08.011.

Abstract

Nucleic acids are powerful triggers of innate immunity and can adopt the Z-conformation, an unusual left-handed double helix. Here, we studied the biological function(s) of Z-RNA recognition by the adenosine deaminase ADAR1, mutations in which cause Aicardi-Goutières syndrome. Adar1mZα/mZα mice, bearing two point mutations in the Z-nucleic acid binding (Zα) domain that abolish Z-RNA binding, displayed spontaneous induction of type I interferons (IFNs) in multiple organs, including in the lung, where both stromal and hematopoietic cells showed IFN-stimulated gene (ISG) induction. Lung neutrophils expressed ISGs induced by the transcription factor IRF3, indicating an initiating role for neutrophils in this IFN response. The IFN response in Adar1mZα/mZα mice required the adaptor MAVS, implicating cytosolic RNA sensing. Adenosine-to-inosine changes were enriched in transposable elements and revealed a specific requirement of ADAR1's Zα domain in editing of a subset of RNAs. Thus, endogenous RNAs in Z-conformation have immunostimulatory potential curtailed by ADAR1, with relevance to autoinflammatory disease in humans.

Keywords: ADAR1; Aicardi–Goutières syndrome; MAVS; MDA5; RNA editing; Z-RNA; Zα domain; influenza A virus; interferon; neutrophil.

Publication types

  • Research Support, Non-U.S. Gov't

MeSH terms

  • Adaptor Proteins, Signal Transducing / immunology*
  • Adenosine / genetics
  • Adenosine / metabolism
  • Adenosine Deaminase / genetics*
  • Animals
  • Autoimmune Diseases of the Nervous System / genetics
  • Autoimmune Diseases of the Nervous System / immunology
  • Inosine / genetics
  • Inosine / metabolism
  • Interferon Type I / genetics
  • Interferon Type I / immunology*
  • Mice
  • Mutation
  • Nervous System Malformations / genetics
  • Nervous System Malformations / immunology
  • RNA Editing / genetics
  • RNA, Double-Stranded / genetics*
  • RNA, Double-Stranded / metabolism

Substances

  • Adaptor Proteins, Signal Transducing
  • IPS-1 protein, mouse
  • Interferon Type I
  • RNA, Double-Stranded
  • Inosine
  • ADAR1 protein, mouse
  • Adenosine Deaminase
  • Adenosine

Supplementary concepts

  • Aicardi-Goutieres syndrome