{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,5,1]],"date-time":"2026-05-01T05:02:08Z","timestamp":1777611728642,"version":"3.51.4"},"reference-count":65,"publisher":"Public Library of Science (PLoS)","issue":"2","license":[{"start":{"date-parts":[[2024,2,27]],"date-time":"2024-02-27T00:00:00Z","timestamp":1708992000000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/creativecommons.org\/licenses\/by\/4.0\/"}],"funder":[{"name":"NIH","award":["R01 DC014932"],"award-info":[{"award-number":["R01 DC014932"]}]},{"name":"NIH","award":["R00 AG062746"],"award-info":[{"award-number":["R00 AG062746"]}]},{"name":"CPRIT Scholar in Cancer Research","award":["RR200063"],"award-info":[{"award-number":["RR200063"]}]},{"name":"NSF","award":["R00AG062746"],"award-info":[{"award-number":["R00AG062746"]}]},{"name":"Baylor College of Medicine Intellectual and Developmental Disabilities Research Center","award":["U54HD083092"],"award-info":[{"award-number":["U54HD083092"]}]}],"content-domain":{"domain":["www.plosone.org"],"crossmark-restriction":false},"short-container-title":["PLoS ONE"],"abstract":"<jats:p>Johnston\u2019s organ, the <jats:italic>Drosophila<\/jats:italic> auditory organ, is anatomically very different from the mammalian organ of Corti. However, recent evidence indicates significant cellular and molecular similarities exist between vertebrate and invertebrate hearing, suggesting that <jats:italic>Drosophila<\/jats:italic> may be a useful platform to determine the function of the many mammalian deafness genes whose underlying biological mechanisms are poorly characterized. Our goal was a comprehensive screen of all known orthologues of mammalian deafness genes in the fruit fly to better understand conservation of hearing mechanisms between the insect and the fly and ultimately gain insight into human hereditary deafness. We used bioinformatic comparisons to screen previously reported human and mouse deafness genes and found that 156 of them have orthologues in <jats:italic>Drosophila melanogaster<\/jats:italic>. We used fluorescent imaging of T2A-GAL4 gene trap and GFP or YFP fluorescent protein trap lines for 54 of the <jats:italic>Drosophila<\/jats:italic> genes and found 38 to be expressed in different cell types in Johnston\u2019s organ. We phenotypically characterized the function of strong loss-of-function mutants in three genes expressed in Johnston\u2019s organ (<jats:italic>Cad99C<\/jats:italic>, <jats:italic>Msp-300<\/jats:italic>, and <jats:italic>Koi<\/jats:italic>) using a courtship assay and electrophysiological recordings of sound-evoked potentials. <jats:italic>Cad99C<\/jats:italic> and <jats:italic>Koi<\/jats:italic> were found to have significant courtship defects. However, when we tested these genes for electrophysiological defects in hearing response, we did not see a significant difference suggesting the courtship defects were not caused by hearing deficiencies. Furthermore, we used a UAS\/RNAi approach to test the function of seven genes and found two additional genes, <jats:italic>CG5921<\/jats:italic> and <jats:italic>Myo10a<\/jats:italic>, that gave a statistically significant delay in courtship but not in sound-evoked potentials. Our results suggest that many mammalian deafness genes have <jats:italic>Drosophila<\/jats:italic> homologues expressed in the Johnston\u2019s organ, but that their requirement for hearing may not necessarily be the same as in mammals.<\/jats:p>","DOI":"10.1371\/journal.pone.0297846","type":"journal-article","created":{"date-parts":[[2024,2,27]],"date-time":"2024-02-27T18:22:50Z","timestamp":1709058170000},"page":"e0297846","update-policy":"https:\/\/doi.org\/10.1371\/journal.pone.corrections_policy","source":"Crossref","is-referenced-by-count":1,"title":["Comparative exploration of mammalian deafness gene homologues in the Drosophila auditory organ shows genetic correlation between insect and vertebrate hearing"],"prefix":"10.1371","volume":"19","author":[{"given":"Daniel C.","family":"Sutton","sequence":"first","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-3086-7225","authenticated-orcid":true,"given":"Jonathan 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