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rdf:type |
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lifeskim:mentions |
umls-concept:C0011065,
umls-concept:C0015745,
umls-concept:C0332453,
umls-concept:C0344315,
umls-concept:C0443281,
umls-concept:C0599894,
umls-concept:C0678226,
umls-concept:C1274040,
umls-concept:C1332102,
umls-concept:C1415470,
umls-concept:C1521840
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pubmed:issue |
8
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pubmed:dateCreated |
2002-4-24
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pubmed:abstractText |
HAP-1 is a huntingtin-associated protein that is enriched in the brain. To gain insight into the normal physiological role of HAP-1, mice were generated with homozygous disruption at the Hap1 locus. Loss of HAP-1 expression did not alter the gross brain expression levels of its interacting partners, huntingtin and p150glued. Newborn Hap1(-/-) animals are observed at the expected Mendelian frequency suggesting a non-essential role of HAP-1 during embryogenesis. Postnatally, Hap1(-/-) pups show decreased feeding behavior that ultimately leads to malnutrition, dehydration and premature death. Seventy percent of Hap1(-/-) pups fail to survive past the second postnatal day (P2) and 100% of Hap1(-/-) pups fail to survive past P9. From P2 until death, Hap1(-/-) pups show markedly decreased amounts of ingested milk. Hap1(-/-) pups that survive to P8 show signs of starvation including greatly decreased serum leptin levels, decreased brain weight and atrophy of the brain cortical mantel. HAP-1 is particularly enriched in the hypothalamus, which is well documented to regulate feeding behavior. Our results demonstrate that HAP-1 plays an essential role in regulating postnatal feeding.
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pubmed:language |
eng
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pubmed:journal |
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pubmed:citationSubset |
IM
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pubmed:chemical |
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pubmed:status |
MEDLINE
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pubmed:month |
Apr
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pubmed:issn |
0964-6906
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pubmed:author |
pubmed-author:ChanEdmond Y WEY,
pubmed-author:ColemanSarahS,
pubmed-author:GertsensteinMarinaM,
pubmed-author:GutekunstClaire-AnneCA,
pubmed-author:HaydenMichael RMR,
pubmed-author:MaasAlexA,
pubmed-author:MacleanAlanA,
pubmed-author:MetzlerMartinaM,
pubmed-author:NagyAndràsA,
pubmed-author:NasirJamalJ,
pubmed-author:RossChristopher ACA
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pubmed:issnType |
Print
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pubmed:day |
15
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pubmed:volume |
11
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pubmed:owner |
NLM
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pubmed:authorsComplete |
Y
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pubmed:pagination |
945-59
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pubmed:dateRevised |
2008-11-21
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pubmed:meshHeading |
pubmed-meshheading:11971876-Animal Nutritional Physiological Phenomena,
pubmed-meshheading:11971876-Animals,
pubmed-meshheading:11971876-Animals, Suckling,
pubmed-meshheading:11971876-Body Constitution,
pubmed-meshheading:11971876-Eating,
pubmed-meshheading:11971876-Homozygote,
pubmed-meshheading:11971876-Hypothalamus,
pubmed-meshheading:11971876-Leptin,
pubmed-meshheading:11971876-Mice,
pubmed-meshheading:11971876-Microtubule-Associated Proteins,
pubmed-meshheading:11971876-Mutagenesis, Site-Directed,
pubmed-meshheading:11971876-Nerve Tissue Proteins,
pubmed-meshheading:11971876-Neurons,
pubmed-meshheading:11971876-Nuclear Proteins,
pubmed-meshheading:11971876-Starvation
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pubmed:year |
2002
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pubmed:articleTitle |
Targeted disruption of Huntingtin-associated protein-1 (Hap1) results in postnatal death due to depressed feeding behavior.
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pubmed:affiliation |
Center for Molecular Medicine and Therapeutics, Department of Medical Genetics, Children's and Women's Hospital, University of British Columbia, Vancouver, British Columbia, Canada.
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pubmed:publicationType |
Journal Article,
Research Support, U.S. Gov't, Non-P.H.S.,
Research Support, Non-U.S. Gov't
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