Rapid functional genetics of the oligodendrocyte lineage using pluripotent stem cells

Nat Commun. 2018 Sep 13;9(1):3708. doi: 10.1038/s41467-018-06102-7.


Oligodendrocyte dysfunction underlies many neurological disorders, but rapid assessment of mutation-specific effects in these cells has been impractical. To enable functional genetics in oligodendrocytes, here we report a highly efficient method for generating oligodendrocytes and their progenitors from mouse embryonic and induced pluripotent stem cells, independent of mouse strain or mutational status. We demonstrate that this approach, when combined with genome engineering, provides a powerful platform for the expeditious study of genotype-phenotype relationships in oligodendrocytes.

Publication types

  • Research Support, N.I.H., Extramural
  • Research Support, Non-U.S. Gov't

MeSH terms

  • Alleles
  • Animals
  • CRISPR-Cas Systems
  • Cell Differentiation / genetics
  • Cell Lineage*
  • DNA Mutational Analysis
  • Genetic Association Studies
  • Genetic Engineering
  • Genotype
  • Induced Pluripotent Stem Cells
  • Lentivirus
  • Male
  • Mice
  • Mice, Inbred C57BL
  • Mice, Knockout
  • Neurons / metabolism
  • Oligodendroglia / cytology*
  • Pluripotent Stem Cells / cytology*