Passenger Mutations Confound Phenotypes of SARM1-Deficient Mice

Cell Rep. 2020 Apr 7;31(1):107498. doi: 10.1016/j.celrep.2020.03.062.

Abstract

The Toll/IL-1R-domain-containing adaptor protein SARM1 is expressed primarily in the brain, where it mediates axonal degeneration. Roles for SARM1 in TLR signaling, viral infection, inflammasome activation, and chemokine and Xaf1 expression have also been described. Much of the evidence for SARM1 function relies on SARM1-deficient mice generated in 129 ESCs and backcrossed to B6. The Sarm1 gene lies in a gene-rich region encompassing Xaf1 and chemokine loci, which remain 129 in sequence. We therefore generated additional knockout strains on the B6 background, confirming the role of SARM1 in axonal degeneration and WNV infection, but not in VSV or LACV infection, or in chemokine or Xaf1 expression. Sequence variation in proapoptotic Xaf1 between B6 and 129 results in coding changes and distinct splice variants, which may account for phenotypes previously attributed to SARM1. Reevaluation of phenotypes in these strains will be critical for understanding the function of SARM1.

Keywords: SARM1; TLR signaling; Wallerian degeneration; Xaf1; chemokines; viral infection.

Publication types

  • Research Support, N.I.H., Extramural

MeSH terms

  • Adaptor Proteins, Signal Transducing / metabolism
  • Animals
  • Apoptosis / genetics
  • Apoptosis Regulatory Proteins / metabolism
  • Armadillo Domain Proteins / genetics*
  • Armadillo Domain Proteins / metabolism*
  • Axons / metabolism
  • Brain / metabolism
  • Cytoskeletal Proteins / genetics*
  • Cytoskeletal Proteins / metabolism*
  • Encephalitis, California / genetics
  • Eye Diseases, Hereditary
  • Female
  • La Crosse virus
  • Male
  • Mice
  • Mice, 129 Strain
  • Mice, Inbred C57BL
  • Mice, Knockout
  • Mutation / genetics
  • Phenotype
  • Receptors, Interleukin-1 / metabolism
  • Retinal Degeneration
  • Signal Transduction / genetics
  • Toll-Like Receptors / metabolism
  • Tumor Necrosis Factor-alpha / metabolism
  • Vision Disorders
  • West Nile Fever / genetics

Substances

  • Adaptor Proteins, Signal Transducing
  • Apoptosis Regulatory Proteins
  • Armadillo Domain Proteins
  • Cytoskeletal Proteins
  • Receptors, Interleukin-1
  • SARM1 protein, mouse
  • Toll-Like Receptors
  • Tumor Necrosis Factor-alpha
  • XAF1 protein, mouse

Supplementary concepts

  • Enhanced S-Cone Syndrome