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Cell Dev. Biol."],"abstract":"<jats:p>Meniere\u2019s disease (MD) is an inner ear disorder characterised by recurrent vertigo attacks associated with sensorineural hearing loss and tinnitus. Evidence from epidemiology and Whole Exome Sequencing (WES) suggests a genetic susceptibility involving multiple genes, including \u03b1-Dystrobrevin (<jats:italic>DTNA<\/jats:italic>). Here we investigate a <jats:italic>Drosophila<\/jats:italic> model. We show that mutation, or knockdown, of the <jats:italic>DTNA<\/jats:italic> orthologue in <jats:italic>Drosophila<\/jats:italic>, <jats:italic>Dystrobrevin<\/jats:italic> (<jats:italic>Dyb<\/jats:italic>), results in defective proprioception and impaired function of Johnston\u2019s Organ (JO), the fly\u2019s equivalent of the inner ear. <jats:italic>Dyb<\/jats:italic> and another component of the dystrophin-glycoprotein complex (DGC), <jats:italic>Dystrophin<\/jats:italic> (<jats:italic>Dys<\/jats:italic>), are expressed in support cells within JO. Their specific locations suggest that they form part of support cell contacts, thereby helping to maintain the integrity of the hemolymph-neuron diffusion barrier, which is equivalent to a blood-brain barrier. These results have important implications for the human condition, and notably, we note that <jats:italic>DTNA<\/jats:italic> is expressed in equivalent cells of the mammalian inner ear.<\/jats:p>","DOI":"10.3389\/fcell.2022.1015651","type":"journal-article","created":{"date-parts":[[2022,11,10]],"date-time":"2022-11-10T17:25:29Z","timestamp":1668101129000},"update-policy":"https:\/\/doi.org\/10.3389\/crossmark-policy","source":"Crossref","is-referenced-by-count":8,"title":["A Drosophila model for Meniere\u2019s disease: Dystrobrevin is required for support cell function in hearing and proprioception"],"prefix":"10.3389","volume":"10","author":[{"given":"T.","family":"Requena","sequence":"first","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"A.","family":"Keder","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"P.","family":"zur Lage","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"J. 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