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Spinocerebellar Ataxia Type 1 Characteristics in Patient-Derived Fibroblast and iPSC-Derived Neuronal Cultures.

Ronald A M BuijsenMichel HuMaria Sáez-GonzálezSofia NotopoulouEleni MinaWinette KoningSarah L GardinerLinda M van der GraafElena DaoutsaliBarry A PepersHailiang MeiVera van DisJean-Philippe FrimatArn M J M van den MaagdenbergSpyros PetrakisWilleke M C van Roon-Mom
Published in: Movement disorders : official journal of the Movement Disorder Society (2023)
Patient-derived cells recapitulate key pathological features of SCA1 pathogenesis providing a valuable tool for the identification of novel disease-specific processes. This model can be used for high throughput screenings to identify compounds, which may prevent or rescue neurodegeneration in this devastating disease. © 2023 The Authors. Movement Disorders published by Wiley Periodicals LLC on behalf of International Parkinson and Movement Disorder Society.
Keyphrases
  • high throughput
  • induced apoptosis
  • early onset
  • cell cycle arrest
  • systematic review
  • oxidative stress
  • randomized controlled trial
  • signaling pathway
  • brain injury
  • bioinformatics analysis