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It has generally been believed that the primary role of the kinocilium and cilia-associated genes in hair cells is in the establishment of the polarity of actin-based stereocilia, the hair cell mechanotransduction apparatus. Through genetic screening and testing of candidate genes in zebrafish (Danio rerio) we have found that mutations in multiple cilia genes implicated in intraflagellar transport (dync2h1, wdr35, ift88, and traf3ip), and the ciliary transition zone (cc2d2a, mks1, and cep290) lead to resistance to aminoglycoside-induced hair cell death. These genes appear to have differing roles in hair cells, as mutations in intraflagellar transport genes, but not transition zone genes, lead to defects in kinocilia formation and processes dependent upon hair cell mechanotransduction activity. These mutants highlight a novel role of cilia-associated genes in hair cells, and provide powerful tools for further study.<\/jats:p>","DOI":"10.1534\/g3.116.030080","type":"journal-article","created":{"date-parts":[[2016,8,1]],"date-time":"2016-08-01T22:38:41Z","timestamp":1470091121000},"page":"2225-2235","source":"Crossref","is-referenced-by-count":25,"title":["Cilia-Associated Genes Play Differing Roles in Aminoglycoside-Induced Hair Cell Death in Zebrafish"],"prefix":"10.1093","volume":"6","author":[{"given":"Tamara M","family":"Stawicki","sequence":"first","affiliation":[{"name":"Department of Biological Structure, University of Washington, Seattle, Washington 98195"},{"name":"Virginia Merrill Bloedel Hearing Research Center, University of Washington, Seattle, Washington 98195"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Liana","family":"Hernandez","sequence":"additional","affiliation":[{"name":"Department of Biological Structure, University of Washington, Seattle, Washington 98195"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Robert","family":"Esterberg","sequence":"additional","affiliation":[{"name":"Virginia Merrill Bloedel Hearing Research Center, University of Washington, Seattle, Washington 98195"},{"name":"Department of Otolaryngology, Head and Neck Surgery, University of Washington, Seattle, Washington, 98195"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Tor","family":"Linbo","sequence":"additional","affiliation":[{"name":"Department of Biological Structure, University of Washington, Seattle, Washington 98195"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Kelly N","family":"Owens","sequence":"additional","affiliation":[{"name":"Department of Biological Structure, University of Washington, Seattle, Washington 98195"},{"name":"Virginia Merrill Bloedel Hearing Research Center, University of Washington, Seattle, Washington 98195"},{"name":"Department of Otolaryngology, Head and Neck Surgery, University of Washington, Seattle, Washington, 98195"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Arish N","family":"Shah","sequence":"additional","affiliation":[{"name":"Division of Basic Sciences, Fred Hutchinson Cancer Research Center, Seattle, Washington, 98109"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Nihal","family":"Thapa","sequence":"additional","affiliation":[{"name":"Department of Biological Structure, University of Washington, Seattle, Washington 98195"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Brock","family":"Roberts","sequence":"additional","affiliation":[{"name":"Department of Biological Structure, University of Washington, Seattle, Washington 98195"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Cecilia B","family":"Moens","sequence":"additional","affiliation":[{"name":"Division of Basic Sciences, Fred Hutchinson Cancer Research Center, Seattle, Washington, 98109"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Edwin W","family":"Rubel","sequence":"additional","affiliation":[{"name":"Virginia Merrill Bloedel Hearing Research Center, University of Washington, Seattle, Washington 98195"},{"name":"Department of Otolaryngology, Head and Neck Surgery, University of Washington, Seattle, Washington, 98195"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"David W","family":"Raible","sequence":"additional","affiliation":[{"name":"Department of Biological Structure, University of Washington, Seattle, Washington 98195"},{"name":"Department of Biological Structure, University of Washington, Seattle, Washington 98195"},{"name":"Virginia Merrill Bloedel Hearing Research Center, University of Washington, Seattle, Washington 98195"}],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"286","published-online":{"date-parts":[[2016,7,1]]},"reference":[{"key":"2021042105451036700_bib1","doi-asserted-by":"crossref","first-page":"113","DOI":"10.1080\/13816810701537424","article-title":"The retinal ciliopathies.","volume":"28","author":"Adams","year":"2007","journal-title":"Ophthalmic Genet."},{"key":"2021042105451036700_bib2","doi-asserted-by":"crossref","first-page":"e22347","DOI":"10.1371\/journal.pone.0022347","article-title":"Functional hair cell mechanotransducer channels are required for aminoglycoside ototoxicity.","volume":"6","author":"Alharazneh","year":"2011","journal-title":"PLoS One"},{"key":"2021042105451036700_bib3","doi-asserted-by":"crossref","first-page":"4041","DOI":"10.1093\/hmg\/ddr332","article-title":"The ciliopathy gene cc2d2a controls zebrafish photoreceptor outer segment development through a role in Rab8-dependent vesicle trafficking.","volume":"20","author":"Bachmann-Gagescu","year":"2011","journal-title":"Hum. 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