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Here, we show that mutation of the mouse orthologue of<jats:italic>GPSM2<\/jats:italic>affects actin-rich stereocilia elongation in auditory and vestibular hair cells, causing deafness and balance defects. The G-protein subunit G\u03b1<jats:sub>i3<\/jats:sub>, a well-documented partner of Gpsm2, participates in the elongation process, and its absence also causes hearing deficits. We show that Gpsm2 defines an \u223c200\u2009nm nanodomain at the tips of stereocilia and this localization requires the presence of G\u03b1<jats:sub>i3<\/jats:sub>, myosin 15 and whirlin. Using single-molecule tracking, we report that loss of<jats:italic>Gpsm2<\/jats:italic>leads to decreased outgrowth and a disruption of actin dynamics in neuronal growth cones. Our results elucidate the aetiology of CMCS and highlight a new molecular role for Gpsm2\/G\u03b1<jats:sub>i3<\/jats:sub>in the regulation of actin dynamics in epithelial and neuronal tissues.<\/jats:p>","DOI":"10.1038\/ncomms14907","type":"journal-article","created":{"date-parts":[[2017,4,7]],"date-time":"2017-04-07T09:37:24Z","timestamp":1491557844000},"update-policy":"https:\/\/doi.org\/10.1007\/springer_crossmark_policy","source":"Crossref","is-referenced-by-count":97,"title":["Defective Gpsm2\/G\u03b1i3 signalling disrupts stereocilia development and growth cone actin dynamics in Chudley-McCullough syndrome"],"prefix":"10.1038","volume":"8","author":[{"given":"Stephanie A.","family":"Mauriac","sequence":"first","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Yeri E.","family":"Hien","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Jonathan E.","family":"Bird","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Steve Dos-Santos","family":"Carvalho","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Ronan","family":"Peyroutou","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Sze Chim","family":"Lee","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Maite M.","family":"Moreau","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Jean-Michel","family":"Blanc","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Aysegul","family":"Gezer","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Chantal","family":"Medina","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Olivier","family":"Thoumine","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Sandra","family":"Beer-Hammer","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Thomas B.","family":"Friedman","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Lukas","family":"R\u00fcttiger","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Andrew","family":"Forge","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Bernd","family":"N\u00fcrnberg","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Nathalie","family":"Sans","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-8739-6519","authenticated-orcid":false,"given":"Mireille","family":"Montcouquiol","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"297","published-online":{"date-parts":[[2017,4,7]]},"reference":[{"key":"BFncomms14907_CR1","doi-asserted-by":"crossref","first-page":"350","DOI":"10.1002\/(SICI)1096-8628(19970131)68:3<350::AID-AJMG19>3.0.CO;2-S","volume":"68","author":"AE Chudley","year":"1997","unstructured":"Chudley, A. 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