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Mutations of MYO7A are responsible for abnormal shaping of hair bundles, resulting in human deafness and murine deafness\/circling behavior. Myo7aa, expressed in SHCs of the inner ear and lateral line of zebrafish, causes <jats:italic>circling<\/jats:italic> behavior and abnormal hair cell function when deficient in <jats:italic>mariner<\/jats:italic> mutant. This work identifies a new hair cell\u2010specific enhancer, highly conserved between species, located within Intron 2\u20133 of zebrafish myosin 7a (<jats:italic>myo7aa<\/jats:italic>) gene. This enhancer is contained within a 761\u2010bp DNA fragment that encompasses a newly identified Exon of <jats:italic>myo7aa<\/jats:italic> and whose activity does not depend on orientation. Compensation of <jats:italic>mariner<\/jats:italic> mutation by expression of mCherry\u2010Myo7aa fusion protein under the control of this 761\u2010bp DNA fragment results in recovery of balance, normal hair bundle shape and restored hair cell function. Two smaller adjacent fragments (344\u2010bp and 431\u2010bp), extracted from the 761\u2010bp fragment, both show hair cell\u2010specific enhancing activity, with apparently reduced intensity and coverage. These data should help understand the role of Myo7aa in sensory hair cell differentiation and function. They provide tools to decipher how <jats:italic>myo7aa<\/jats:italic> gene is expressed and regulated in SHCs by allowing the identification of potential transcription factors involved in this process. The discovered enhancer could represent a new target for the identification of deafness\u2010causing mutations affecting human <jats:italic>MYO7A<\/jats:italic>. \u00a9 2015 Wiley Periodicals, Inc. Develop Neurobiol 75: 961\u2013983, 2015<\/jats:p>","DOI":"10.1002\/dneu.22263","type":"journal-article","created":{"date-parts":[[2014,12,30]],"date-time":"2014-12-30T07:55:19Z","timestamp":1419926119000},"page":"961-983","source":"Crossref","is-referenced-by-count":7,"title":["A genomic region encompassing a newly identified exon provides enhancing activity sufficient for normal myo7aa expression in zebrafish sensory hair cells"],"prefix":"10.1002","volume":"75","author":[{"given":"Sylvain","family":"Ernest","sequence":"first","affiliation":[{"name":"INSERM U 1024 CNRS UMR 8197 IBENS: Institut de Biologie de l'Ecole Normale Sup\u00e9rieure 75005 Paris France"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Fr\u00e9d\u00e9ric M.","family":"Rosa","sequence":"additional","affiliation":[{"name":"INSERM U 1024 CNRS UMR 8197 IBENS: Institut de Biologie de l'Ecole Normale Sup\u00e9rieure 75005 Paris France"}],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"311","published-online":{"date-parts":[[2015,1,19]]},"reference":[{"key":"e_1_2_6_2_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.ydbio.2011.03.007"},{"key":"e_1_2_6_3_1","doi-asserted-by":"publisher","DOI":"10.1387\/ijdb.072422ba"},{"key":"e_1_2_6_4_1","doi-asserted-by":"publisher","DOI":"10.1002\/cne.1308"},{"key":"e_1_2_6_5_1","doi-asserted-by":"publisher","DOI":"10.1126\/science.284.5421.1837"},{"key":"e_1_2_6_6_1","doi-asserted-by":"publisher","DOI":"10.1101\/gr.133546.111"},{"key":"e_1_2_6_7_1","doi-asserted-by":"publisher","DOI":"10.1093\/hmg\/10.15.1581"},{"key":"e_1_2_6_8_1","doi-asserted-by":"publisher","DOI":"10.1097\/WCO.0b013e32834ef8b2"},{"key":"e_1_2_6_9_1","doi-asserted-by":"publisher","DOI":"10.1038\/cr.2013.45"},{"key":"e_1_2_6_10_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.modgep.2006.09.004"},{"key":"e_1_2_6_11_1","doi-asserted-by":"publisher","DOI":"10.1093\/hmg\/9.14.2189"},{"key":"e_1_2_6_12_1","first-page":"291","volume-title":"Functional Vertebrate Morphology","author":"Fay RR","year":"1985"},{"key":"e_1_2_6_13_1","doi-asserted-by":"publisher","DOI":"10.1101\/gad.1568407"},{"key":"e_1_2_6_14_1","doi-asserted-by":"publisher","DOI":"10.1038\/374062a0"},{"key":"e_1_2_6_15_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.devcel.2006.02.019"},{"key":"e_1_2_6_16_1","doi-asserted-by":"publisher","DOI":"10.1242\/dev.125.23.4637"},{"key":"e_1_2_6_17_1","doi-asserted-by":"publisher","DOI":"10.1002\/(SICI)1096-9861(19960129)365:1<113::AID-CNE9>3.0.CO;2-6"},{"key":"e_1_2_6_18_1","doi-asserted-by":"publisher","DOI":"10.1023\/A:1007013904913"},{"key":"e_1_2_6_19_1","doi-asserted-by":"publisher","DOI":"10.1083\/jcb.137.6.1287"},{"key":"e_1_2_6_20_1","doi-asserted-by":"publisher","DOI":"10.1038\/nbt.2501"},{"key":"e_1_2_6_21_1","doi-asserted-by":"publisher","DOI":"10.1074\/jbc.M210489200"},{"key":"e_1_2_6_22_1","doi-asserted-by":"publisher","DOI":"10.1016\/S1534-5807(02)00409-4"},{"key":"e_1_2_6_23_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.devcel.2004.06.005"},{"key":"e_1_2_6_24_1","doi-asserted-by":"publisher","DOI":"10.1002\/aja.1002030302"},{"key":"e_1_2_6_25_1","doi-asserted-by":"publisher","DOI":"10.1038\/nature12298"},{"key":"e_1_2_6_26_1","doi-asserted-by":"publisher","DOI":"10.1006\/geno.2000.6451"},{"key":"e_1_2_6_27_1","doi-asserted-by":"publisher","DOI":"10.1242\/dev.02734"},{"key":"e_1_2_6_28_1","doi-asserted-by":"publisher","DOI":"10.1016\/S0896-6273(00)80455-9"},{"key":"e_1_2_6_29_1","doi-asserted-by":"publisher","DOI":"10.1146\/annurev.genet.39.073003.105049"},{"key":"e_1_2_6_30_1","doi-asserted-by":"publisher","DOI":"10.1002\/(SICI)1098-1004(199910)14:4<354::AID-HUMU18>3.0.CO;2-Z"},{"key":"e_1_2_6_31_1","doi-asserted-by":"publisher","DOI":"10.1038\/ng1296-385"},{"key":"e_1_2_6_32_1","doi-asserted-by":"publisher","DOI":"10.1038\/ng.2426"},{"key":"e_1_2_6_33_1","doi-asserted-by":"publisher","DOI":"10.1006\/dbio.1997.8736"},{"key":"e_1_2_6_34_1","doi-asserted-by":"publisher","DOI":"10.1101\/gr.078519.108"},{"key":"e_1_2_6_35_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.ydbio.2006.03.037"},{"key":"e_1_2_6_36_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.heares.2012.11.001"},{"key":"e_1_2_6_37_1","doi-asserted-by":"publisher","DOI":"10.1002\/(SICI)1097-4695(19991115)41:3<424::AID-NEU10>3.0.CO;2-G"},{"key":"e_1_2_6_38_1","doi-asserted-by":"publisher","DOI":"10.1126\/science.1243490"},{"key":"e_1_2_6_39_1","doi-asserted-by":"publisher","DOI":"10.1074\/jbc.M111.228304"},{"key":"e_1_2_6_40_1","doi-asserted-by":"publisher","DOI":"10.1523\/JNEUROSCI.5554-10.2011"},{"key":"e_1_2_6_41_1","doi-asserted-by":"publisher","DOI":"10.1038\/374060a0"},{"key":"e_1_2_6_42_1","doi-asserted-by":"publisher","DOI":"10.1038\/ng0697-191"},{"key":"e_1_2_6_43_1","volume-title":"The Zebrafish Book. 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