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Gipc1 has a dual role in Vangl2 trafficking and hair bundle integrity in the inner ear

dc.rights.licenseopenen_US
hal.structure.identifierNeurocentre Magendie : Physiopathologie de la Plasticité Neuronale [U1215 Inserm - UB]
dc.contributor.authorGIESE, Arnaud P.
hal.structure.identifierNeurocentre Magendie : Physiopathologie de la Plasticité Neuronale [U1215 Inserm - UB]
dc.contributor.authorEZAN, Jerome
dc.contributor.authorWANG, Lingyan
hal.structure.identifierNeurocentre Magendie : Physiopathologie de la Plasticité Neuronale [U1215 Inserm - UB]
dc.contributor.authorLASVAUX, Lea
dc.contributor.authorLEMBO, Frederique
hal.structure.identifierNeurocentre Magendie : Physiopathologie de la Plasticité Neuronale [U1215 Inserm - UB]
dc.contributor.authorMAZZOCCO, Claire
hal.structure.identifierNeurocentre Magendie : Physiopathologie de la Plasticité Neuronale [U1215 Inserm - UB]
dc.contributor.authorRICHARD, Elodie
dc.contributor.authorREBOUL, Jerome
dc.contributor.authorBORG, Jean-Paul
dc.contributor.authorKELLEY, Matthew W.
hal.structure.identifierNeurocentre Magendie : Physiopathologie de la Plasticité Neuronale [U1215 Inserm - UB]
dc.contributor.authorSANS, Nathalie
dc.contributor.authorBRIGANDE, John
hal.structure.identifierNeurocentre Magendie : Physiopathologie de la Plasticité Neuronale [U1215 Inserm - UB]
dc.contributor.authorMONTCOUQUIOL, Mireille
dc.date.accessioned2022-06-30T13:09:51Z
dc.date.available2022-06-30T13:09:51Z
dc.date.issued2012-10-15
dc.identifier.issn1477-9129en_US
dc.identifier.urihttp://www.ncbi.nlm.nih.gov/pubmed/22991442
dc.identifier.urioadoi:http://europepmc.org/articles/pmc4067280
dc.identifier.urioadoi:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC4067280/pdf
dc.identifier.urioai:crossref.org:10.1242/dev.074229
dc.identifier.uriftpubmed:oai:pubmedcentral.nih.gov:4067280
dc.identifier.urioadoi:http://www.ncbi.nlm.nih.gov/pmc/articles/PMC4067280
dc.identifier.urihttps://oskar-bordeaux.fr/handle/20.500.12278/140345
dc.description.abstractEnVangl2 is one of the central proteins controlling the establishment of planar cell polarity in multiple tissues of different species. Previous studies suggest that the localization of the Vangl2 protein to specific intracellular microdomains is crucial for its function. However, the molecular mechanisms that control Vangl2 trafficking within a cell are largely unknown. Here, we identify Gipc1 (GAIP C-terminus interacting protein 1) as a new interactor for Vangl2, and we show that a myosin VI-Gipc1 protein complex can regulate Vangl2 traffic in heterologous cells. Furthermore, we show that in the cochlea of MyoVI mutant mice, Vangl2 presence at the membrane is increased, and that a disruption of Gipc1 function in hair cells leads to maturation defects, including defects in hair bundle orientation and integrity. Finally, stimulated emission depletion microscopy and overexpression of GFP-Vangl2 show an enrichment of Vangl2 on the supporting cell side, adjacent to the proximal membrane of hair cells. Altogether, these results indicate a broad role for Gipc1 in the development of both stereociliary bundles and cell polarization, and suggest that the strong asymmetry of Vangl2 observed in early postnatal cochlear epithelium is mostly a ‘tissue’ polarity readout.
dc.description.sponsorshipCaractérisation moléculaire et fonctionnelle de vangl2 et de ses partenaires chez le mammifèreen_US
dc.description.sponsorshipCaractérisations moléculaire et fonctionnelle de la protéine Scribble 1 au cours du développement du système nerveux central - ANR-07-NEUR-0031en_US
dc.language.isoENen_US
dc.sourceoadoi_repo
dc.sourcecrossref
dc.sourcebase
dc.subject.enPolarity
dc.subject.enTraffic
dc.subject.enActin
dc.subject.enCochlea
dc.subject.enVan Gogh-like 2
dc.subject.enGipc1
dc.subject.enMyosin VI
dc.subject.enPlanar polarity
dc.subject.enMouse
dc.subject.enRat
dc.title.enGipc1 has a dual role in Vangl2 trafficking and hair bundle integrity in the inner ear
dc.typeArticle de revueen_US
dc.identifier.doi10.1242/dev.074229en_US
dc.subject.halSciences du Vivant [q-bio]/Neurosciences [q-bio.NC]en_US
dc.identifier.pubmed22991442en_US
bordeaux.journalDevelopment (Cambridge, England)en_US
bordeaux.page3775-3785en_US
bordeaux.volume139en_US
bordeaux.hal.laboratoriesNeurocentre Magendie - UMR-S 1215en_US
bordeaux.issue20en_US
bordeaux.institutionUniversité de Bordeauxen_US
bordeaux.institutionINSERMen_US
bordeaux.peerReviewedouien_US
bordeaux.inpressnonen_US
bordeaux.identifier.funderIDConseil Régional Aquitaineen_US
bordeaux.identifier.funderIDFondation pour la Recherche Médicaleen_US
bordeaux.identifier.funderIDEuropean Commissionen_US
bordeaux.identifier.funderIDNational Institute on Deafness and Other Communication Disordersen_US
bordeaux.identifier.funderIDLigue Contre le Canceren_US
bordeaux.import.sourcedissemin
hal.identifierhal-03710271
hal.version1
hal.date.transferred2022-06-30T13:10:00Z
hal.exporttrue
workflow.import.sourcedissemin
dc.rights.ccPas de Licence CCen_US
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