Nature Communications (Dec 2022)

Huntington disease oligodendrocyte maturation deficits revealed by single-nucleus RNAseq are rescued by thiamine-biotin supplementation

  • Ryan G. Lim,
  • Osama Al-Dalahmah,
  • Jie Wu,
  • Maxwell P. Gold,
  • Jack C. Reidling,
  • Guomei Tang,
  • Miriam Adam,
  • David K. Dansu,
  • Hye-Jin Park,
  • Patrizia Casaccia,
  • Ricardo Miramontes,
  • Andrea M. Reyes-Ortiz,
  • Alice Lau,
  • Richard A. Hickman,
  • Fatima Khan,
  • Fahad Paryani,
  • Alice Tang,
  • Kenneth Ofori,
  • Emily Miyoshi,
  • Neethu Michael,
  • Nicolette McClure,
  • Xena E. Flowers,
  • Jean Paul Vonsattel,
  • Shawn Davidson,
  • Vilas Menon,
  • Vivek Swarup,
  • Ernest Fraenkel,
  • James E. Goldman,
  • Leslie M. Thompson

DOI
https://doi.org/10.1038/s41467-022-35388-x
Journal volume & issue
Vol. 13, no. 1
pp. 1 – 23

Abstract

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Here the authors evaluate single cell gene expression from mouse and human Huntington’s disease brains, finding incomplete oligodendrocyte maturation and pathways involved. Treating mice with thiamine/biotin ameliorates molecular pathology.