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VCP recruitment to mitochondria causes mitophagy impairment and neurodegeneration in models of Huntington's disease
Xing Guo, Xiaoyan Sun, Di Hu, Ya Wang Juan, Hisashi Fujioka, , Sudha Chakrapani, Amit Joshi Umesh, Yu Luo, Daria Mochly-RosenShow More
Published in
2016
Volume: 7
   
Issue: 1
Pages: 1 - 17
Abstract
Mutant Huntingtin (mtHtt) causes neurodegeneration in Huntington's disease (HD) by evoking defects in the mitochondria, but the underlying mechanisms remains elusive. Our proteomic analysis identifies valosin-containing protein (VCP) as an mtHtt-binding protein on the mitochondria. Here we show that VCP is selectively translocated to the mitochondria, where it is bound to mtHtt in various HD models. Mitochondria-accumulated VCP elicits excessive mitophagy, causing neuronal cell death. Blocking mtHtt/VCP mitochondrial interaction with a peptide, HV-3, abolishes VCP translocation to the mitochondria, corrects excessive mitophagy and reduces cell death in HD mouse- and patient-derived cells and HD transgenic mouse brains. Treatment with HV-3 reduces behavioural and neuropathological phenotypes of HD in both fragment- and full-length mtHtt transgenic mice. Our findings demonstrate a causal role of mtHtt-induced VCP mitochondrial accumulation in HD pathogenesis and suggest that the peptide HV-3 might be a useful tool for developing new therapeutics to treat HD.
About the journal
Published in
Open Access
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