Establishment of DYT5 patient-specific induced pluripotent stem cells with a GCH1 mutation

Stem Cell Res. 2017 Oct:24:36-39. doi: 10.1016/j.scr.2017.07.029. Epub 2017 Jul 29.

Abstract

Peripheral blood mononuclear cells (PBMCs) were collected from a clinically diagnosed 20-year-old dystonia patient with a GCH1 mutation (DYT5). Episomal vectors were used to introduce reprogramming factors (OCT3/4, SOX2, KLF4, L-MYC, LIN28, and p53 carboxy-terminal dominant-negative fragment) to the PBMCs. The generated iPSCs expressed pluripotency markers, and were capable of differentiating into derivates of all three germ layers in vitro. The iPSC line also showed a normal karyotype and preserved the GCH1 mutation. This cellular model can provide opportunities to perform pathophysiological studies for aberrant dopamine metabolism-related disorders.

Publication types

  • Case Reports

MeSH terms

  • Adult
  • Cell Differentiation
  • Genetic Vectors / genetics*
  • Humans
  • Induced Pluripotent Stem Cells / metabolism*
  • Kruppel-Like Factor 4
  • Male
  • Mutation
  • Transcription Factors / genetics
  • Young Adult

Substances

  • KLF4 protein, human
  • Kruppel-Like Factor 4
  • Transcription Factors