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Though studied extensively, the photoreceptor-specific roles of CEP290 remain unclear. Using advanced microscopy techniques, we investigated the sub-ciliary localization of CEP290 and its role in mouse photoreceptors during development. CEP290 was found throughout the connecting cilium between the microtubules and membrane, with nine-fold symmetry. In the absence of CEP290 ciliogenesis occurs, but the connecting cilium membrane is aberrant, and sub-structures, such as the ciliary necklace and Y-links, are defective or absent throughout the mid to distal connecting cilium. Transition zone proteins AHI1 and NPHP1 were abnormally restricted to the proximal connecting cilium in the absence of CEP290, while others like NPHP8 and CEP89 were unaffected. Although outer segment disc formation is inhibited in CEP290 mutant retina, we observed large numbers of extracellular vesicles. These results suggest roles for CEP290 in ciliary membrane structure, outer segment disc formation and photoreceptor-specific spatial distribution of a subset of transition zone proteins, which collectively lead to failure of outer segment formation and photoreceptor degeneration.\n                <\/jats:p>","DOI":"10.1101\/2025.01.20.633784","type":"posted-content","created":{"date-parts":[[2025,1,20]],"date-time":"2025-01-20T23:45:15Z","timestamp":1737416715000},"source":"Crossref","is-referenced-by-count":1,"title":["Ciliopathy-associated protein, CEP290, is required for ciliary necklace and outer segment membrane formation in retinal photoreceptors"],"prefix":"10.64898","author":[{"given":"Abigail R.","family":"Moye","sequence":"first","affiliation":[{"name":"Institute of Molecular and Clinical Ophthalmology Basel (IOB), Basel, 4031, Switzerland"},{"name":"Department of Ophthalmology, University of Basel, Basel, 4031, Switzerland"},{"name":"Verna and Marrs McLean Department of Biochemistry and Molecular Biology, Baylor College of Medicine, Houston, TX, USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Michael A.","family":"Robichaux","sequence":"additional","affiliation":[{"name":"Department of Ophthalmology & Visual Sciences and Department of Biochemistry & Molecular Medicine, West Virginia University, Morgantown, West Virginia, USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Melina A.","family":"Agosto","sequence":"additional","affiliation":[{"name":"Retina and Optic Nerve Research Laboratory, Department of Physiology and Biophysics, and Department of Ophthalmology and Visual Sciences, Dalhousie University, Halifax, Nova Scotia, Canada"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-0733-9950","authenticated-orcid":false,"given":"Carlo","family":"Rivolta","sequence":"additional","affiliation":[{"name":"Institute of Molecular and Clinical Ophthalmology Basel (IOB), Basel, 4031, Switzerland"},{"name":"Department of Ophthalmology, University of Basel, Basel, 4031, Switzerland"},{"name":"Verna and Marrs McLean Department of Biochemistry and Molecular Biology, Baylor College of Medicine, Houston, TX, USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Alexandre P.","family":"Moulin","sequence":"additional","affiliation":[{"name":"Jules-Gonin Eye Hospital, Fondation Asile des Aveugles, University of Lausanne, 1004 Lausanne, Switzerland"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Theodore G.","family":"Wensel","sequence":"additional","affiliation":[{"name":"Verna and Marrs McLean Department of Biochemistry and Molecular Biology, Baylor College of Medicine, Houston, TX, USA"}],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"54368","reference":[{"key":"2026041021575888000_2025.01.20.633784v1.1","doi-asserted-by":"publisher","DOI":"10.1517\/14728222.2012.680956"},{"key":"2026041021575888000_2025.01.20.633784v1.2","doi-asserted-by":"publisher","DOI":"10.1038\/ng2069"},{"key":"2026041021575888000_2025.01.20.633784v1.3","doi-asserted-by":"publisher","DOI":"10.1038\/s41467-020-14767-2"},{"key":"2026041021575888000_2025.01.20.633784v1.4","doi-asserted-by":"publisher","DOI":"10.1086\/519494"},{"key":"2026041021575888000_2025.01.20.633784v1.5","doi-asserted-by":"publisher","DOI":"10.1093\/hmg\/ddu738"},{"key":"2026041021575888000_2025.01.20.633784v1.6","doi-asserted-by":"publisher","DOI":"10.1002\/humu.22618"},{"key":"2026041021575888000_2025.01.20.633784v1.7","doi-asserted-by":"publisher","DOI":"10.1371\/journal.pone.0207958"},{"key":"2026041021575888000_2025.01.20.633784v1.8","doi-asserted-by":"publisher","DOI":"10.1086\/519026"},{"key":"2026041021575888000_2025.01.20.633784v1.9","doi-asserted-by":"publisher","DOI":"10.1016\/j.ophtha.2018.05.026"},{"key":"2026041021575888000_2025.01.20.633784v1.10","doi-asserted-by":"publisher","DOI":"10.1083\/jcb.19.1.79"},{"key":"2026041021575888000_2025.01.20.633784v1.11","doi-asserted-by":"publisher","DOI":"10.1093\/hmg\/ddl107"},{"key":"2026041021575888000_2025.01.20.633784v1.12","doi-asserted-by":"publisher","DOI":"10.1016\/j.semcdb.2020.07.013"},{"key":"2026041021575888000_2025.01.20.633784v1.13","doi-asserted-by":"crossref","unstructured":"Chen, Q. , Zhao, H. , Pan, X. , Fang, C. , Qiu, B. , Guo, J. , Yan, X. & Zhu, X . 2024. 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