Hap1

Summary

Gene Symbol: Hap1
Description: huntingtin-associated protein 1
Alias: HAP-1, huntingtin-associated protein 1
Species: mouse
Products:     Hap1

Top Publications

  1. Dragatsis I, Dietrich P, Zeitlin S. Expression of the Huntingtin-associated protein 1 gene in the developing and adult mouse. Neurosci Lett. 2000;282:37-40 pubmed
    Huntingtin-associated protein 1 (HAP1) interacts with the product of the Huntington's disease gene...
  2. Lin Y, Xu X, Cape A, Li S, Li X. Huntingtin-associated protein-1 deficiency in orexin-producing neurons impairs neuronal process extension and leads to abnormal behavior in mice. J Biol Chem. 2010;285:15941-9 pubmed publisher
    Huntingtin-associated protein-1 (Hap1) is a neuronal protein that associates with huntingtin, the Huntington disease protein...
  3. McGuire J, Rong J, Li S, Li X. Interaction of Huntingtin-associated protein-1 with kinesin light chain: implications in intracellular trafficking in neurons. J Biol Chem. 2006;281:3552-9 pubmed
    Huntingtin-associated protein-1 (HAP1) was initially identified as an interacting partner of huntingtin, the Huntington disease protein...
  4. Zucker B, Luthi Carter R, Kama J, Dunah A, Stern E, Fox J, et al. Transcriptional dysregulation in striatal projection- and interneurons in a mouse model of Huntington's disease: neuronal selectivity and potential neuroprotective role of HAP1. Hum Mol Genet. 2005;14:179-89 pubmed
    ..NMDA receptor subunit NR2D, the postsynaptic density protein 95 (PSD-95) and the huntingtin-associated protein 1 (HAP1) as well as nitric-oxide-synthase (nNOS) mRNA itself...
  5. Li S, Yu Z, Li C, Nguyen H, Zhou Y, Deng C, et al. Lack of huntingtin-associated protein-1 causes neuronal death resembling hypothalamic degeneration in Huntington's disease. J Neurosci. 2003;23:6956-64 pubmed
    ..Here, we show that huntingtin-associated protein-1 (HAP1), which is involved in intracellular trafficking of epidermal growth factor receptor (EGFR), is highly expressed in ..
  6. Bertaux F, Sharp A, Ross C, Lehrach H, Bates G, Wanker E. HAP1-huntingtin interactions do not contribute to the molecular pathology in Huntington's disease transgenic mice. FEBS Lett. 1998;426:229-32 pubmed
    b>HAP1 (huntingtin associated protein) has previously been found to interact with huntingtin (htt) in a glutamine length dependent manner and has been proposed to play a role in the cell specific neurodegeneration observed in Huntington's ..
  7. Wu L, Fan Y, Li S, Li X, Zhou X. Huntingtin-associated protein-1 interacts with pro-brain-derived neurotrophic factor and mediates its transport and release. J Biol Chem. 2010;285:5614-23 pubmed publisher
    ..Here, we report that Huntingtin-associated protein-1 (HAP1) interacts with the prodomain of BDNF and that the interaction was reduced in the presence of polyQ-expanded ..
  8. Sheng G, Xu X, Lin Y, Wang C, Rong J, Cheng D, et al. Huntingtin-associated protein 1 interacts with Ahi1 to regulate cerebellar and brainstem development in mice. J Clin Invest. 2008;118:2785-95 pubmed publisher
    ..In this study, we found that mouse Ahi1 formed a stable complex with huntingtin-associated protein 1 (Hap1), which is critical for neonatal development and involved in intracellular trafficking...
  9. Chan E, Nasir J, Gutekunst C, Coleman S, Maclean A, Maas A, et al. Targeted disruption of Huntingtin-associated protein-1 (Hap1) results in postnatal death due to depressed feeding behavior. Hum Mol Genet. 2002;11:945-59 pubmed
    ..To gain insight into the normal physiological role of HAP-1, mice were generated with homozygous disruption at the Hap1 locus...

More Information

Publications39

  1. Gauthier L, Charrin B, Borrell Pages M, Dompierre J, Rangone H, Cordelières F, et al. Huntingtin controls neurotrophic support and survival of neurons by enhancing BDNF vesicular transport along microtubules. Cell. 2004;118:127-38 pubmed
    ..Huntingtin-mediated transport involves huntingtin-associated protein-1 (HAP1) and the p150(Glued) subunit of dynactin, an essential component of molecular motors...
  2. Yang G, Yang M, Lim Y, Lu J, Wang T, Qi J, et al. Huntingtin associated protein 1 regulates trafficking of the amyloid precursor protein and modulates amyloid beta levels in neurons. J Neurochem. 2012;122:1010-22 pubmed publisher
    ..However, the mechanism of APP trafficking remains unclear. We present evidence that huntingtin associated protein 1 (HAP1) may reduce A? production by regulating APP trafficking to the non-amyloidogenic pathway...
  3. Nasir J, Duan K, Nichol K, Engelender S, Ashworth R, Colomer V, et al. Gene structure and map location of the murine homolog of the Huntington-associated protein, Hap1. Mamm Genome. 1998;9:565-70 pubmed
    ..We have mapped its murine homolog, Hap1, to mouse Chr 11 (band D), which shares extensive synteny with human Chr 17 including the region 17q21-q22, where ..
  4. Sheng G, Chang G, Lin J, Yu Z, Fang Z, Rong J, et al. Hypothalamic huntingtin-associated protein 1 as a mediator of feeding behavior. Nat Med. 2006;12:526-33 pubmed
    ..Huntingtin-associated protein 1 (Hap1) is expressed more abundantly in the hypothalamus than in other brain regions, and lack of Hap1 in mice leads to ..
  5. Martin E, Kim M, Velier J, Sapp E, Lee H, Laforet G, et al. Analysis of Huntingtin-associated protein 1 in mouse brain and immortalized striatal neurons. J Comp Neurol. 1999;403:421-30 pubmed
    ..Results support a role for HAP-1 in vesicle trafficking and organelle movement in mitotic cells and differentiated neurons and implicate HAP-1B as the predominant molecular subtype associated with vesicle membranes in striatal neurons. ..
  6. Mackenzie K, Lim Y, Duffield M, Chataway T, Zhou X, Keating D. Huntingtin-associated protein-1 (HAP1) regulates endocytosis and interacts with multiple trafficking-related proteins. Cell Signal. 2017;35:176-187 pubmed publisher
    Huntingtin-associated protein 1 (HAP1) was initially identified as a binding partner of huntingtin, mutations in which underlie Huntington's disease...
  7. Niu S, Huang Z, Wang H, Rao X, Kong H, Xu J, et al. Brainstem Hap1-Ahi1 is involved in insulin-mediated feeding control. FEBS Lett. 2011;585:85-91 pubmed publisher
    The function of the brainstem Hap1-Ahi1 complex in the regulation of feeding behavior was investigated. When mice were fasted or treated with 2-deoxy-D-glucose (2-DG), Hap1-Ahi1 was significantly upregulated...
  8. Mackenzie K, Duffield M, Peiris H, Phillips L, Zanin M, Teo E, et al. Huntingtin-associated protein 1 regulates exocytosis, vesicle docking, readily releasable pool size and fusion pore stability in mouse chromaffin cells. J Physiol. 2014;592:1505-18 pubmed publisher
    Huntingtin-associated protein 1 (HAP1) was initially established as a neuronal binding partner of huntingtin, mutations in which underlie Huntington's disease...
  9. Nasir J, MacLean A, Engelender S, Duan K, Margolis R, Kleiderlein J, et al. Chromosomal localization of the Huntingtin associated protein (HAP-1) gene in mouse and humans with radiation hybrid and interspecific backcross mapping. Mamm Genome. 1999;10:397-8 pubmed
  10. Rong J, Li S, Sheng G, Wu M, Coblitz B, Li M, et al. 14-3-3 protein interacts with Huntingtin-associated protein 1 and regulates its trafficking. J Biol Chem. 2007;282:4748-56 pubmed
    b>HAP1 (Huntingtin-associated protein 1) consists of two alternately spliced isoforms (HAP1A and HAP1B, which have unique C-terminal sequences) and participates in intracellular trafficking...
  11. Xiang J, Yan S, Li S, Li X. Postnatal loss of hap1 reduces hippocampal neurogenesis and causes adult depressive-like behavior in mice. PLoS Genet. 2015;11:e1005175 pubmed publisher
    ..Here we genetically depleted the expression of huntingtin-associated protein 1 (Hap1) in mice at various ages or in selective brain regions...
  12. Huang P, Chen C, Hsu I, Salim S, Kao S, Cheng C, et al. Huntingtin-associated protein 1 interacts with breakpoint cluster region protein to regulate neuronal differentiation. PLoS ONE. 2015;10:e0116372 pubmed publisher
    ..Huntingtin (Htt)-associated protein-1 (Hap1) is a brain-enriched protein and plays a key role in the trafficking of neuronal surviving and differentiating ..
  13. Xiang J, Yang S, Xin N, Gaertig M, Reeves R, Li S, et al. DYRK1A regulates Hap1-Dcaf7/WDR68 binding with implication for delayed growth in Down syndrome. Proc Natl Acad Sci U S A. 2017;114:E1224-E1233 pubmed publisher
    Huntingtin-associated protein 1 (Hap1) is known to be critical for postnatal hypothalamic function and growth...
  14. Tang T, Tu H, Orban P, Chan E, Hayden M, Bezprozvanny I. HAP1 facilitates effects of mutant huntingtin on inositol 1,4,5-trisphosphate-induced Ca release in primary culture of striatal medium spiny neurons. Eur J Neurosci. 2004;20:1779-87 pubmed
    Huntington's disease is caused by polyglutamine expansion (exp) in huntingtin (Htt). Htt-associated protein-1 (HAP1) was the first identified Htt-binding partner...
  15. Cape A, Chen X, Wang C, O Neill A, Lin Y, He J, et al. Loss of huntingtin-associated protein 1 impairs insulin secretion from pancreatic ?-cells. Cell Mol Life Sci. 2012;69:1305-17 pubmed publisher
    b>Hap1 was originally identified as a neuronal protein that interacts with huntingtin, the Huntington's disease (HD) protein...
  16. Nasir J, Lafuente M, Duan K, Colomer V, Engelender S, Ingersoll R, et al. Human huntingtin-associated protein (HAP-1) gene: genomic organisation and an intragenic polymorphism. Gene. 2000;254:181-7 pubmed
    ..The detailed characterisation of the genomic organisation of HAP-1 and the presence of an intragenic polymorphism will be helpful in evaluating its role in different disorders, using candidate gene approaches. ..
  17. Fulp C, Cho G, Marsh E, Nasrallah I, Labosky P, Golden J. Identification of Arx transcriptional targets in the developing basal forebrain. Hum Mol Genet. 2008;17:3740-60 pubmed publisher
  18. Keryer G, Pineda J, Liot G, Kim J, Dietrich P, Benstaali C, et al. Ciliogenesis is regulated by a huntingtin-HAP1-PCM1 pathway and is altered in Huntington disease. J Clin Invest. 2011;121:4372-82 pubmed publisher
    ..Here, we demonstrate that WT HTT regulates ciliogenesis by interacting through huntingtin-associated protein 1 (HAP1) with pericentriolar material 1 protein (PCM1)...
  19. Yang M, Lim Y, Li X, Zhong J, Zhou X. Precursor of brain-derived neurotrophic factor (proBDNF) forms a complex with Huntingtin-associated protein-1 (HAP1) and sortilin that modulates proBDNF trafficking, degradation, and processing. J Biol Chem. 2011;286:16272-84 pubmed publisher
    ..In this study, we report that proBDNF forms a complex with Huntingtin associated protein-1 (HAP1) and sortilin, which plays an important role in proBDNF intracellular trafficking and stabilization...
  20. Mejia L, Litterman N, Ikeuchi Y, de la Torre Ubieta L, Bennett E, Zhang C, et al. A novel Hap1-Tsc1 interaction regulates neuronal mTORC1 signaling and morphogenesis in the brain. J Neurosci. 2013;33:18015-21 pubmed publisher
    ..approach in neural cells including neurons, we uncover the brain-enriched protein huntingtin-associated protein 1 (Hap1) as a novel functional partner of Tsc1...
  21. Pan J, Yuan S, Yu T, Su C, Liu X, He J, et al. Regulation of L-type Ca2+ Channel Activity and Insulin Secretion by Huntingtin-associated Protein 1. J Biol Chem. 2016;291:26352-26363 pubmed
    Huntingtin-associated protein 1 (Hap1) was originally identified as a protein that binds to the Huntington disease protein, huntingtin...
  22. Xiang J, Yang H, Zhao T, Sun M, Xu X, Zhou X, et al. Huntingtin-associated protein 1 regulates postnatal neurogenesis and neurotrophin receptor sorting. J Clin Invest. 2014;124:85-98 pubmed publisher
    ..Huntingtin-associated protein 1 (HAP1) participates in intracellular trafficking in neurons, and its absence leads to postnatal death in mice...
  23. Dragatsis I, Efstratiadis A, Zeitlin S. Mouse mutant embryos lacking huntingtin are rescued from lethality by wild-type extraembryonic tissues. Development. 1998;125:1529-39 pubmed
  24. Mackenzie K, Lumsden A, Guo F, Duffield M, Chataway T, Lim Y, et al. Huntingtin-associated protein-1 is a synapsin I-binding protein regulating synaptic vesicle exocytosis and synapsin I trafficking. J Neurochem. 2016;138:710-21 pubmed publisher
    Huntingtin-associated protein-1 (HAP1) is involved in intracellular trafficking, vesicle transport, and membrane receptor endocytosis...
  25. Dragatsis I, Zeitlin S, Dietrich P. Huntingtin-associated protein 1 (Hap1) mutant mice bypassing the early postnatal lethality are neuroanatomically normal and fertile but display growth retardation. Hum Mol Genet. 2004;13:3115-25 pubmed
    Huntingtin-associated protein 1 (Hap1) is the first huntingtin interacting protein identified in a yeast two-hybrid screen...
  26. Tuz K, Hsiao Y, Ju√°rez O, Shi B, Harmon E, Phelps I, et al. The Joubert syndrome-associated missense mutation (V443D) in the Abelson-helper integration site 1 (AHI1) protein alters its localization and protein-protein interactions. J Biol Chem. 2013;288:13676-94 pubmed publisher
    ..Mutations in AHI1 decrease cilia formation, alter its localization and stability, and change its binding to HAP1 and NPHP1. Mutations in AHI1 affect ciliogenesis, AHI1 protein localization, and AHI1-protein interactions...
  27. Wong Y, Holzbaur E. The regulation of autophagosome dynamics by huntingtin and HAP1 is disrupted by expression of mutant huntingtin, leading to defective cargo degradation. J Neurosci. 2014;34:1293-305 pubmed publisher
    ..Here, we show that both huntingtin (htt) and its adaptor protein huntingtin-associated protein-1 (HAP1) copurify and colocalize with autophagosomes in neurons...
  28. Prigge J, Schmidt E. HAP1 can sequester a subset of TBP in cytoplasmic inclusions via specific interaction with the conserved TBP(CORE). BMC Mol Biol. 2007;8:76 pubmed
    ..Htt-associated protein 1 (HAP1), a component of neuronal cytoplasmic stigmoid bodies (STBs), can sequester polyQ-expanded Htt and AR in STBs, ..
  29. Kim H, Ryu J, Woo H, Cho S, Sung M, Kim S, et al. Patterns of gene expression associated with Pten deficiency in the developing inner ear. PLoS ONE. 2014;9:e97544 pubmed publisher
    ..From this study, we suggest two key regulatory signaling networks mediated by Spp1 and Rgs4, which may play potential roles in neuronal differentiation of developing auditory neurons. ..
  30. Rui Y, Xu Z, Patel B, Chen Z, Chen D, Tito A, et al. Huntingtin functions as a scaffold for selective macroautophagy. Nat Cell Biol. 2015;17:262-75 pubmed publisher
    ..Our data uncover an important physiological function of Huntingtin and provide a missing link in the activation of selective macroautophagy in metazoans. ..