{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,1,20]],"date-time":"2026-01-20T09:10:44Z","timestamp":1768900244650,"version":"3.49.0"},"reference-count":90,"publisher":"Public Library of Science (PLoS)","issue":"3","license":[{"start":{"date-parts":[[2022,3,14]],"date-time":"2022-03-14T00:00:00Z","timestamp":1647216000000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/creativecommons.org\/licenses\/by\/4.0\/"}],"funder":[{"DOI":"10.13039\/100000053","name":"National Eye Institute","doi-asserted-by":"publisher","award":["T32 EY024234"],"award-info":[{"award-number":["T32 EY024234"]}],"id":[{"id":"10.13039\/100000053","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/100000053","name":"National Eye Institute","doi-asserted-by":"publisher","award":["F31 EY030758"],"award-info":[{"award-number":["F31 EY030758"]}],"id":[{"id":"10.13039\/100000053","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/100000053","name":"National Eye Institute","doi-asserted-by":"publisher","award":["R01 EY025378"],"award-info":[{"award-number":["R01 EY025378"]}],"id":[{"id":"10.13039\/100000053","id-type":"DOI","asserted-by":"publisher"}]}],"content-domain":{"domain":["www.plosone.org"],"crossmark-restriction":false},"short-container-title":["PLoS ONE"],"abstract":"<jats:p>Cilia are essential for the development and function of many different tissues. Although cilia machinery is crucial in the eye for photoreceptor development and function, a role for cilia in early eye development and morphogenesis is still somewhat unclear: many zebrafish cilia mutants retain cilia at early stages due to maternal deposition of cilia components. An eye phenotype has been described in the mouse <jats:italic>Arl13<\/jats:italic> mutant, however, zebrafish <jats:italic>arl13b<\/jats:italic> is maternally deposited, and an early role for cilia proteins has not been tested in zebrafish eye development. Here we use the zebrafish <jats:italic>dzip1<\/jats:italic> mutant, which exhibits a loss of cilia throughout stages of early eye development, to examine eye development and morphogenesis. We find that in <jats:italic>dzip1<\/jats:italic> mutants, initial formation of the optic cup proceeds normally, however, the optic fissure subsequently fails to close and embryos develop the structural eye malformation ocular coloboma. Further, neural crest cells, which are implicated in optic fissure closure, do not populate the optic fissure correctly, suggesting that their inappropriate localization may be the underlying cause of coloboma. Overall, our results indicate a role for <jats:italic>dzip1<\/jats:italic> in proper neural crest localization in the optic fissure and optic fissure closure.<\/jats:p>","DOI":"10.1371\/journal.pone.0265327","type":"journal-article","created":{"date-parts":[[2022,3,14]],"date-time":"2022-03-14T17:36:44Z","timestamp":1647279404000},"page":"e0265327","update-policy":"https:\/\/doi.org\/10.1371\/journal.pone.corrections_policy","source":"Crossref","is-referenced-by-count":4,"title":["Loss of zebrafish dzip1 results in inappropriate recruitment of periocular mesenchyme to the optic fissure and ocular coloboma"],"prefix":"10.1371","volume":"17","author":[{"given":"Sri Pratima","family":"Nandamuri","sequence":"first","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-0602-4391","authenticated-orcid":true,"given":"Sarah","family":"Lusk","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-0052-275X","authenticated-orcid":true,"given":"Kristen M.","family":"Kwan","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"340","published-online":{"date-parts":[[2022,3,14]]},"reference":[{"issue":"1","key":"pone.0265327.ref001","doi-asserted-by":"crossref","first-page":"1","DOI":"10.1016\/j.ydbio.2007.01.045","article-title":"Molecular mechanisms of optic vesicle development: complexities, ambiguities and controversies","volume":"305","author":"R Adler","year":"2007","journal-title":"Dev Biol"},{"key":"pone.0265327.ref002","doi-asserted-by":"crossref","first-page":"73","DOI":"10.1016\/j.gde.2015.02.004","article-title":"Watching eyes take shape","volume":"32","author":"N Bazin-Lopez","year":"2015","journal-title":"Curr Opin Genet Dev"},{"key":"pone.0265327.ref003","doi-asserted-by":"crossref","first-page":"128","DOI":"10.1016\/j.ydbio.2021.03.023","article-title":"Build me up optic cup: Intrinsic and extrinsic mechanisms of vertebrate eye morphogenesis","volume":"476","author":"MA Casey","year":"2021","journal-title":"Dev Biol"},{"key":"pone.0265327.ref004","doi-asserted-by":"crossref","first-page":"255","DOI":"10.1146\/annurev.cellbio.17.1.255","article-title":"Early eye development in vertebrates","volume":"17","author":"RL Chow","year":"2001","journal-title":"Annu Rev Cell Dev Biol"},{"key":"pone.0265327.ref005","doi-asserted-by":"crossref","first-page":"61","DOI":"10.1016\/B978-0-12-385044-7.00003-5","article-title":"Eye morphogenesis and patterning of the optic vesicle","volume":"93","author":"S. 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