論文

査読有り
2010年5月

Mutant huntingtin impairs Ku70-mediated DNA repair

JOURNAL OF CELL BIOLOGY
  • Yasushi Enokido
  • Takuya Tamura
  • Hikaru Ito
  • Anup Arumughan
  • Akihiko Komuro
  • Hiroki Shiwaku
  • Masaki Sone
  • Raphaele Foulle
  • Hirohide Sawada
  • Hiroshi Ishiguro
  • Tetsuya Ono
  • Miho Murata
  • Ichiro Kanazawa
  • Nikolai Tomilin
  • Kazuhiko Tagawa
  • Erich E. Wanker
  • Hitoshi Okazawa
  • 全て表示

189
3
開始ページ
425
終了ページ
443
記述言語
英語
掲載種別
研究論文(学術雑誌)
DOI
10.1083/jcb.200905138
出版者・発行元
ROCKEFELLER UNIV PRESS

DNA repair defends against naturally occurring or disease-associated DNA damage during the long lifespan of neurons and is implicated in polyglutamine disease pathology. In this study, we report that mutant huntingtin (Htt) expression in neurons causes double-strand breaks (DSBs) of genomic DNA, and Htt further promotes DSBs by impairing DNA repair. We identify Ku70, a component of the DNA damage repair complex, as a mediator of the DNA repair dysfunction in mutant Htt-expressing neurons. Mutant Htt interacts with Ku70, impairs DNA-dependent protein kinase function in nonhomologous end joining, and consequently increases DSB accumulation. Expression of exogenous Ku70 rescues abnormal behavior and pathological phenotypes in the R6/2 mouse model of Huntington's disease (HD). These results collectively suggest that Ku70 is a critical regulator of DNA damage in HD pathology.

リンク情報
DOI
https://doi.org/10.1083/jcb.200905138
PubMed
https://www.ncbi.nlm.nih.gov/pubmed/20439996
Web of Science
https://gateway.webofknowledge.com/gateway/Gateway.cgi?GWVersion=2&SrcAuth=JSTA_CEL&SrcApp=J_Gate_JST&DestLinkType=FullRecord&KeyUT=WOS:000277269600008&DestApp=WOS_CPL
ID情報
  • DOI : 10.1083/jcb.200905138
  • ISSN : 0021-9525
  • PubMed ID : 20439996
  • Web of Science ID : WOS:000277269600008

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