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Hum Mol Genet. 2011 Oct 15;20(20):3899-909. doi: 10.1093/hmg/ddr308. Epub 2011 Jul 20.

Altered palmitoylation and neuropathological deficits in mice lacking HIP14.

Author information

1
Centre for Molecular Medicine and Therapeutics and Child and Family Research Institute, Vancouver, BC, Canada.

Abstract

Huntingtin interacting protein 14 (HIP14, ZDHHC17) is a huntingtin (HTT) interacting protein with palmitoyl transferase activity. In order to interrogate the function of Hip14, we generated mice with disruption in their Hip14 gene. Hip14-/- mice displayed behavioral, biochemical and neuropathological defects that are reminiscent of Huntington disease (HD). Palmitoylation of other HIP14 substrates, but not Htt, was reduced in the Hip14-/- mice. Hip14 is dysfunctional in the presence of mutant htt in the YAC128 mouse model of HD, suggesting that altered palmitoylation mediated by HIP14 may contribute to HD.

PMID:
21775500
PMCID:
PMC3177655
DOI:
10.1093/hmg/ddr308
[Indexed for MEDLINE]
Free PMC Article

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