Dimerization quality control ensures neuronal development and survival

Science. 2018 Oct 12;362(6411):eaap8236. doi: 10.1126/science.aap8236. Epub 2018 Sep 6.

Abstract

Aberrant complex formation by recurrent interaction modules, such as BTB domains, leucine zippers, or coiled coils, can disrupt signal transduction, yet whether cells detect and eliminate complexes of irregular composition is unknown. By searching for regulators of the BTB family, we discovered a quality control pathway that ensures functional dimerization [dimerization quality control (DQC)]. Key to this network is the E3 ligase SCFFBXL17, which selectively binds and ubiquitylates BTB dimers of aberrant composition to trigger their clearance by proteasomal degradation. Underscoring the physiological importance of DQC, SCFFBXL17 is required for the differentiation, function, and survival of neural crest and neuronal cells. We conclude that metazoan organisms actively monitor BTB dimerization, and we predict that distinct E3 ligases similarly control complex formation by other recurrent domains.

Publication types

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

MeSH terms

  • Animals
  • BTB-POZ Domain*
  • Cell Survival
  • F-Box Proteins / genetics
  • F-Box Proteins / metabolism*
  • Humans
  • Mutation
  • Neural Crest / cytology
  • Neural Crest / embryology
  • Neurogenesis*
  • Neurons / physiology*
  • Protein Multimerization*
  • Ubiquitin-Protein Ligases / metabolism*
  • Xenopus laevis

Substances

  • F-Box Proteins
  • FBXL17 protein, human
  • Ubiquitin-Protein Ligases