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Therefore, it is important to diagnose hearing loss soon after birth so that interventions can be provided as early as possible. Most newborns in the United States are screened for hearing deficits and commercially available next\u2010generation sequencing hearing loss panels often can identify the causative gene, which may also identify congenital defects in other organs. One of the most prevalent autosomal dominant congenital hearing loss syndromes is branchio\u2010oto\u2010renal syndrome (BOR), which also presents with defects in craniofacial structures and the kidney. Currently, mutations in three genes,\n                    <jats:italic>SIX1, SIX5<\/jats:italic>\n                    , and\n                    <jats:italic>EYA1<\/jats:italic>\n                    , are known to be causative in about half of the BOR patients that have been tested. To uncover new candidate genes that could be added to congenital hearing loss genetic screens, we have combined the power of\n                    <jats:italic>Drosophila<\/jats:italic>\n                    mutants and protein biochemical assays with the embryological advantages of\n                    <jats:italic>Xenopus<\/jats:italic>\n                    , a key aquatic animal model with a high level of genomic similarity to human, to identify potential Six1 transcriptional targets and interacting proteins that play a role during otic development. We review our transcriptomic, yeast 2\u2010hybrid, and proteomic approaches that have revealed a large number of new candidates. We also discuss how we have begun to identify how Six1 and co\u2010factors interact to direct developmental events necessary for normal otic development.\n                  <\/jats:p>","DOI":"10.1002\/jez.b.23222","type":"journal-article","created":{"date-parts":[[2023,10,13]],"date-time":"2023-10-13T05:14:25Z","timestamp":1697174065000},"page":"212-240","update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":7,"title":["Using\n                    <i>Xenopu<\/i>\n                    s to discover new candidate genes involved in BOR and other congenital hearing loss syndromes"],"prefix":"10.1002","volume":"342","author":[{"given":"Scott J.","family":"Neal","sequence":"first","affiliation":[{"name":"Department of Neuroscience and Physiology Upstate Medical University Syracuse New York 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USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Dominique","family":"Alfandari","sequence":"additional","affiliation":[{"name":"Department of Veterinary and Animal Sciences University of Massachusetts Amherst Amherst Massachusetts USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Francesca","family":"Pignoni","sequence":"additional","affiliation":[{"name":"Department of Neuroscience and Physiology Upstate Medical University Syracuse New York USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-4192-1087","authenticated-orcid":false,"given":"Sally A.","family":"Moody","sequence":"additional","affiliation":[{"name":"Department of Anatomy and Cell Biology George Washington University Washington District of Columbia 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