Fbxo9 functions downstream of Sox10 to determine neuron-glial fate choice in the dorsal root ganglia through Neurog2 destabilization

Proc Natl Acad Sci U S A. 2020 Feb 25;117(8):4199-4210. doi: 10.1073/pnas.1916164117. Epub 2020 Feb 6.

Abstract

The transcription factor Sox10 is a key regulator in the fate determination of a subpopulation of multipotent trunk neural crest (NC) progenitors toward glial cells instead of sensory neurons in the dorsal root ganglia (DRG). However, the mechanism by which Sox10 regulates glial cell fate commitment during lineage segregation remains poorly understood. In our study, we showed that the neurogenic determinant Neurogenin 2 (Neurog2) exhibited transient overlapping expression with Sox10 in avian trunk NC progenitors, which progressively underwent lineage segregation during migration toward the forming DRG. Gain- and loss-of-function studies revealed that the temporary expression of Neurog2 was due to Sox10 regulation of its protein stability. Transcriptional profiling identified Sox10-regulated F-box only protein (Fbxo9), which is an SCF (Skp1-Cul-F-box)-type ubiquitin ligase for Neurog2. Consistently, overexpression of Fbxo9 in NC progenitors down-regulated Neurog2 protein expression through ubiquitination and promoted the glial lineage at the expense of neuronal differentiation, whereas Fbxo9 knockdown resulted in the opposite phenomenon. Mechanistically, we found that Fbxo9 interacted with Neurog2 to promote its destabilization through the F-box motif. Finally, epistasis analysis further demonstrated that Fbxo9 and probably other F-box members mediated the role of Sox10 in destabilizing Neurog2 protein and directing the lineage of NC progenitors toward glial cells rather than sensory neurons. Altogether, these findings unravel a Sox10-Fbxo9 regulatory axis in promoting the glial fate of NC progenitors through Neurog2 destabilization.

Keywords: Fbxo9; Neurog2; Sox10; neural crest progenitors.

Publication types

  • Research Support, Non-U.S. Gov't

MeSH terms

  • Amino Acid Motifs
  • Animals
  • Basic Helix-Loop-Helix Transcription Factors / chemistry
  • Basic Helix-Loop-Helix Transcription Factors / genetics
  • Basic Helix-Loop-Helix Transcription Factors / metabolism*
  • Chick Embryo
  • F-Box Proteins / chemistry
  • F-Box Proteins / genetics
  • F-Box Proteins / metabolism*
  • Female
  • Gene Expression Regulation, Developmental
  • Male
  • Mice
  • Mice, Knockout
  • Nerve Tissue Proteins / chemistry
  • Nerve Tissue Proteins / genetics
  • Nerve Tissue Proteins / metabolism*
  • Neural Crest / cytology
  • Neural Crest / metabolism
  • Neurogenesis
  • Neuroglia / cytology*
  • Neuroglia / metabolism*
  • Neurons / cytology*
  • Neurons / metabolism*
  • Protein Binding
  • Protein Stability
  • SOXE Transcription Factors / metabolism*
  • Spinal Nerve Roots / cytology
  • Spinal Nerve Roots / metabolism*

Substances

  • Basic Helix-Loop-Helix Transcription Factors
  • F-Box Proteins
  • FBXO9 protein, mouse
  • Nerve Tissue Proteins
  • Neurog2 protein, mouse
  • SOXE Transcription Factors
  • Sox10 protein, mouse