Human Neural Stem Cell Transplantation Rescues Functional Deficits in R6/2 and Q140 Huntington's Disease Mice

نویسندگان

  • Jack C. Reidling
  • Aroa Relaño-Ginés
  • Sandra M. Holley
  • Joseph Ochaba
  • Cindy Moore
  • Brian Fury
  • Alice Lau
  • Andrew H. Tran
  • Sylvia Yeung
  • Delaram Salamati
  • Chunni Zhu
  • Asa Hatami
  • Carlos Cepeda
  • Joshua A. Barry
  • Talia Kamdjou
  • Alvin King
  • Dane Coleal-Bergum
  • Nicholas R. Franich
  • Frank M. LaFerla
  • Joan S. Steffan
  • Mathew Blurton-Jones
  • Charles K. Meshul
  • Gerhard Bauer
  • Michael S. Levine
  • Marie-Francoise Chesselet
  • Leslie M. Thompson
چکیده

Huntington's disease (HD) is an inherited neurodegenerative disorder with no disease-modifying treatment. Expansion of the glutamine-encoding repeat in the Huntingtin (HTT) gene causes broad effects that are a challenge for single treatment strategies. Strategies based on human stem cells offer a promising option. We evaluated efficacy of transplanting a good manufacturing practice (GMP)-grade human embryonic stem cell-derived neural stem cell (hNSC) line into striatum of HD modeled mice. In HD fragment model R6/2 mice, transplants improve motor deficits, rescue synaptic alterations, and are contacted by nerve terminals from mouse cells. Furthermore, implanted hNSCs are electrophysiologically active. hNSCs also improved motor and late-stage cognitive impairment in a second HD model, Q140 knockin mice. Disease-modifying activity is suggested by the reduction of aberrant accumulation of mutant HTT protein and expression of brain-derived neurotrophic factor (BDNF) in both models. These findings hold promise for future development of stem cell-based therapies.

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عنوان ژورنال:

دوره 10  شماره 

صفحات  -

تاریخ انتشار 2018