{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,10,17]],"date-time":"2026-10-17T02:20:56Z","timestamp":1792203656874,"version":"4.4.0"},"update-to":[{"DOI":"10.1371\/journal.pgen.1011205","type":"new_version","label":"New version","source":"publisher","updated":{"date-parts":[[2025,3,20]],"date-time":"2025-03-20T00:00:00Z","timestamp":1742428800000}}],"reference-count":69,"publisher":"Public Library of Science (PLoS)","issue":"3","license":[{"start":{"date-parts":[[2025,3,11]],"date-time":"2025-03-11T00:00:00Z","timestamp":1741651200000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/creativecommons.org\/licenses\/by\/4.0\/"}],"funder":[{"DOI":"10.13039\/100000002","name":"National Institutes of Health","doi-asserted-by":"publisher","award":["5R21EY032575"],"award-info":[{"award-number":["5R21EY032575"]}],"id":[{"id":"10.13039\/100000002","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/100001116","name":"Foundation Fighting Blindness","doi-asserted-by":"publisher","award":["TA-NMT-0621-0811-UFL"],"award-info":[{"award-number":["TA-NMT-0621-0811-UFL"]}],"id":[{"id":"10.13039\/100001116","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/100010310","name":"E. Matilda Ziegler Foundation for the Blind","doi-asserted-by":"publisher","id":[{"id":"10.13039\/100010310","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/100001116","name":"Foundation Fighting Blindness","doi-asserted-by":"publisher","award":["PPA-0617-0718"],"award-info":[{"award-number":["PPA-0617-0718"]}],"id":[{"id":"10.13039\/100001116","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/100000002","name":"National Institutes of Health","doi-asserted-by":"publisher","award":["R01 EY026559-06A1"],"award-info":[{"award-number":["R01 EY026559-06A1"]}],"id":[{"id":"10.13039\/100000002","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/100000097","name":"National Center for Research Resources","doi-asserted-by":"crossref","award":["C06RR016511"],"award-info":[{"award-number":["C06RR016511"]}],"id":[{"id":"10.13039\/100000097","id-type":"DOI","asserted-by":"crossref"}]}],"content-domain":{"domain":["www.plosgenetics.org"],"crossmark-restriction":false},"short-container-title":["PLoS Genet"],"abstract":"<jats:p>\n                    Mutations in\n                    <jats:italic>CLRN1<\/jats:italic>\n                    cause Usher syndrome type IIIA (USH3A), an autosomal recessive disorder characterized by hearing and vision loss, and often accompanied by vestibular dysfunction. The identity of the cell types responsible for the pathology and mechanisms leading to vision loss in USH3A remains elusive. To address this, we employed CRISPR\/Cas9 technology to delete a large region in the coding and untranslated (UTR) region of zebrafish\n                    <jats:italic>clrn1<\/jats:italic>\n                    . The retinas of\n                    <jats:italic>clrn1<\/jats:italic>\n                    mutant larvae exhibited sensitivity to cell stress, along with age-dependent loss of function and degeneration in the photoreceptor layer. Investigation revealed disorganization in the outer retina in\n                    <jats:italic>clrn1<\/jats:italic>\n                    mutants, including actin-based structures of the M\u00fcller glia and photoreceptor cells. To assess cell-specific contributions to USH3A pathology, we specifically re-expressed\n                    <jats:italic>clrn1<\/jats:italic>\n                    in either M\u00fcller glia or photoreceptor cells. M\u00fcller glia re-expression of\n                    <jats:italic>clrn1<\/jats:italic>\n                    prevented the elevated cell death observed in larval\n                    <jats:italic>clrn1<\/jats:italic>\n                    mutant zebrafish exposed to high-intensity light. Notably, the degree of phenotypic rescue correlated with the level of Clrn1 re-expression. Surprisingly, high levels of Clrn1 expression enhanced cell death in both wild-type and\n                    <jats:italic>clrn1<\/jats:italic>\n                    mutant animals. However, rod- or cone-specific Clrn1 re-expression did not reduce the extent of cell death. Taken together, our findings underscore three crucial insights. First,\n                    <jats:italic>clrn1<\/jats:italic>\n                    mutant zebrafish exhibit key pathological features of USH3A; second, Clrn1 within M\u00fcller glia plays a pivotal role in photoreceptor maintenance, with its expression requiring controlled regulation; third, the reliance of photoreceptors on M\u00fcller glia suggests a structural support mechanism, possibly through direct interactions between M\u00fcller glia and photoreceptors mediated in part by Clrn1 protein.\n                  <\/jats:p>","DOI":"10.1371\/journal.pgen.1011205","type":"journal-article","created":{"date-parts":[[2025,3,11]],"date-time":"2025-03-11T13:25:22Z","timestamp":1741699522000},"page":"e1011205","update-policy":"https:\/\/doi.org\/10.1371\/journal.pgen.corrections_policy","source":"Crossref","is-referenced-by-count":9,"title":["The USH3A causative gene clarin1 functions in M\u00fcller glia to maintain retinal photoreceptors"],"prefix":"10.1371","volume":"21","author":[{"ORCID":"https:\/\/orcid.org\/0000-0002-6067-8791","authenticated-orcid":true,"given":"Hannah J. T.","family":"Nonarath","sequence":"first","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-6703-9901","authenticated-orcid":true,"given":"Samantha L.","family":"Simpson","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Tricia L.","family":"Slobodianuk","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Hai","family":"Tran","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-1550-299X","authenticated-orcid":true,"given":"Ross F.","family":"Collery","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-6328-8601","authenticated-orcid":true,"given":"Astra","family":"Dinculescu","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-7173-2642","authenticated-orcid":true,"given":"Brian A.","family":"Link","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"340","published-online":{"date-parts":[[2025,3,11]]},"reference":[{"key":"pgen.1011205.ref001","first-page":"827","article-title":"Genetic analysis of Tunisian families with Usher syndrome type 1: toward improving early molecular diagnosis","volume":"22","author":"I Ben-Rebeh","year":"2016","journal-title":"Molecular Vision"},{"issue":"8","key":"pgen.1011205.ref002","doi-asserted-by":"crossref","first-page":"595","DOI":"10.1016\/0021-9681(83)90147-9","article-title":"Usher syndrome: definition and estimate of prevalence from two high-risk populations","volume":"36","author":"JA Boughman","year":"1983","journal-title":"J Chronic Dis"},{"issue":"3","key":"pgen.1011205.ref003","doi-asserted-by":"crossref","first-page":"133","DOI":"10.1016\/0021-9681(69)90055-1","article-title":"Usher\u2019s syndrome--deafness and progressive blindness. 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