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We demonstrate the efficacy of this strategy in mouse USH1F models, effectively restoring hearing and balance in these mice. Importantly, our approach also proves successful in expressing PCDH15 in clinically relevant retinal models, including human retinal organoids and non-human primate retina, showing efficient targeting of photoreceptors and proper protein expression in the calyceal processes. This research represents a major step toward advancing gene therapy for USH1F and the multiple challenges of hearing, balance, and vision impairment.<\/jats:p>","DOI":"10.1101\/2023.11.09.566447","type":"posted-content","created":{"date-parts":[[2023,11,13]],"date-time":"2023-11-13T19:30:15Z","timestamp":1699903815000},"source":"Crossref","is-referenced-by-count":6,"title":["PCDH15 Dual-AAV Gene Therapy for Deafness and Blindness in Usher Syndrome Type 1F"],"prefix":"10.64898","author":[{"ORCID":"https:\/\/orcid.org\/0000-0003-0754-1427","authenticated-orcid":false,"given":"Maryna V.","family":"Ivanchenko","sequence":"first","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-6186-0798","authenticated-orcid":false,"given":"Daniel 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