Huntington's Disease Pathogenesis Is Modified In Vivo by Alfy/Wdfy3 and Selective Macroautophagy

Leora M. Fox, Kiryung Kim, Christopher W. Johnson, Shawei Chen, Katherine R. Croce, Matheus B. Victor, Evelien Eenjes, Joan R. Bosco, Lisa K. Randolph, Ioannis Dragatsis, Joanna M. Dragich, Andrew S. Yoo, Ai Yamamoto

Research output: Contribution to journalArticlepeer-review

19 Scopus citations

Abstract

Fox et al. demonstrate in a Huntington's disease (HD) mouse model and HD-patient-derived neurons that the autophagy adaptor protein Alfy/WDFY3 is required for removal of aggregated protein and that depletion of Alfy hastens the pathogenic onset of HD.

Original languageEnglish
Pages (from-to)813-821.e6
JournalNeuron
Volume105
Issue number5
DOIs
StatePublished - Mar 4 2020

Keywords

  • Alfy
  • Huntington's disease
  • Wdfy3
  • autophagy
  • direct conversion
  • mice
  • neurodegeneration
  • patient fibroblasts
  • proteinopathy
  • selective autophagy

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