Literature DB >> 21775500

Altered palmitoylation and neuropathological deficits in mice lacking HIP14.

Roshni R Singaraja1, Kun Huang, Shaun S Sanders, Austen J Milnerwood, Rochelle Hines, Jason P Lerch, Sonia Franciosi, Renaldo C Drisdel, Kuljeet Vaid, Fiona B Young, Crystal Doty, Junmei Wan, Nagat Bissada, R Mark Henkelman, William N Green, Nicholas G Davis, Lynn A Raymond, Michael R Hayden.   

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.

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Year:  2011        PMID： 21775500      PMCID： PMC3177655          DOI： 10.1093/hmg/ddr308

Source DB:  PubMed          Journal:  Hum Mol Genet        ISSN： 0964-6906            Impact factor:   6.150


  37 in total

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6.  Natural history of disease in the YAC128 mouse reveals a discrete signature of pathology in Huntington disease.

Authors:  Jeffrey B Carroll; Jason P Lerch; Sonia Franciosi; Amanda Spreeuw; Nagat Bissada; R Mark Henkelman; Michael R Hayden
Journal:  Neurobiol Dis       Date:  2011-03-31       Impact factor: 5.996

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Journal:  Hum Mol Genet       Date:  2003-07-01       Impact factor: 6.150

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