{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,3,16]],"date-time":"2026-03-16T16:36:16Z","timestamp":1773678976890,"version":"3.50.1"},"reference-count":85,"publisher":"MDPI AG","issue":"13","license":[{"start":{"date-parts":[[2022,6,27]],"date-time":"2022-06-27T00:00:00Z","timestamp":1656288000000},"content-version":"vor","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0\/"}],"funder":[{"name":"German Research Community DFG","award":["SFB 894"],"award-info":[{"award-number":["SFB 894"]}]},{"name":"German Research Community DFG","award":["HOMFOR"],"award-info":[{"award-number":["HOMFOR"]}]},{"name":"German Research Community DFG","award":["2020-002"],"award-info":[{"award-number":["2020-002"]}]},{"name":"German Research Community DFG","award":["2021-022"],"award-info":[{"award-number":["2021-022"]}]},{"name":"Saarland University","award":["SFB 894"],"award-info":[{"award-number":["SFB 894"]}]},{"name":"Saarland University","award":["HOMFOR"],"award-info":[{"award-number":["HOMFOR"]}]},{"name":"Saarland University","award":["2020-002"],"award-info":[{"award-number":["2020-002"]}]},{"name":"Saarland University","award":["2021-022"],"award-info":[{"award-number":["2021-022"]}]},{"name":"Dr. Rolf M. Schwiete Foundation","award":["SFB 894"],"award-info":[{"award-number":["SFB 894"]}]},{"name":"Dr. Rolf M. Schwiete Foundation","award":["HOMFOR"],"award-info":[{"award-number":["HOMFOR"]}]},{"name":"Dr. Rolf M. Schwiete Foundation","award":["2020-002"],"award-info":[{"award-number":["2020-002"]}]},{"name":"Dr. Rolf M. Schwiete Foundation","award":["2021-022"],"award-info":[{"award-number":["2021-022"]}]}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":["IJMS"],"abstract":"<jats:p>The Unc119 protein mediates transport of myristoylated proteins to the photoreceptor outer segment, a specialized primary cilium. This transport activity is regulated by the GTPase Arl3 as well as by Arl13b and Rp2 that control Arl3 activation\/inactivation. Interestingly, Unc119 is also enriched in photoreceptor synapses and can bind to RIBEYE, the main component of synaptic ribbons. In the present study, we analyzed whether the known regulatory proteins, that control the Unc119-dependent myristoylated protein transport at the primary cilium, are also present at the photoreceptor synaptic ribbon complex by using high-resolution immunofluorescence and immunogold electron microscopy. We found Arl3 and Arl13b to be enriched at the synaptic ribbon whereas Rp2 was predominantly found on vesicles distributed within the entire terminal. These findings indicate that the synaptic ribbon could be involved in the discharge of Unc119-bound lipid-modified proteins. In agreement with this hypothesis, we found Nphp3 (Nephrocystin-3), a myristoylated, Unc119-dependent cargo protein enriched at the basal portion of the ribbon in close vicinity to the active zone. Mutations in Nphp3 are known to be associated with Senior\u2013L\u00f8ken Syndrome 3 (SLS3). Visual impairment and blindness in SLS3 might thus not only result from ciliary dysfunctions but also from malfunctions of the photoreceptor synapse.<\/jats:p>","DOI":"10.3390\/ijms23137135","type":"journal-article","created":{"date-parts":[[2022,6,27]],"date-time":"2022-06-27T22:31:14Z","timestamp":1656369074000},"page":"7135","update-policy":"https:\/\/doi.org\/10.3390\/mdpi_crossmark_policy","source":"Crossref","is-referenced-by-count":12,"title":["Ciliary Proteins Repurposed by the Synaptic Ribbon: Trafficking Myristoylated Proteins at Rod Photoreceptor Synapses"],"prefix":"10.3390","volume":"23","author":[{"given":"Shweta","family":"Suiwal","sequence":"first","affiliation":[{"name":"Institute of Anatomy and Cell Biology, Department of Neuroanatomy, Medical School, Saarland University, 66421 Homburg, Germany"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Mayur","family":"Dembla","sequence":"additional","affiliation":[{"name":"Institute of Anatomy and Cell Biology, Department of Neuroanatomy, Medical School, Saarland University, 66421 Homburg, Germany"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-7328-1427","authenticated-orcid":false,"given":"Karin","family":"Schwarz","sequence":"additional","affiliation":[{"name":"Institute of Anatomy and Cell Biology, Department of Neuroanatomy, Medical School, Saarland University, 66421 Homburg, Germany"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Rashmi","family":"Katiyar","sequence":"additional","affiliation":[{"name":"Institute of Anatomy and Cell Biology, Department of Neuroanatomy, Medical School, Saarland University, 66421 Homburg, Germany"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-1482-7020","authenticated-orcid":false,"given":"Martin","family":"Jung","sequence":"additional","affiliation":[{"name":"Institute of Biochemistry and Molecular Biology, Medical School, Saarland University, 66421 Homburg, Germany"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Yvonne","family":"Carius","sequence":"additional","affiliation":[{"name":"Institute of Biophysics, Department of Structural Biology, Center of Human and Molecular Biology (ZHMB), Medical School, Saarland University, 66421 Homburg, Germany"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-4472-695X","authenticated-orcid":false,"given":"Stephan","family":"Maxeiner","sequence":"additional","affiliation":[{"name":"Institute of Anatomy and Cell Biology, Department of Neuroanatomy, Medical School, Saarland University, 66421 Homburg, Germany"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Marcel A.","family":"Lauterbach","sequence":"additional","affiliation":[{"name":"Molecular Imaging, Center for Integrative Physiology and Molecular Medicine, Medical School, Saarland University, 66421 Homburg, Germany"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"C. Roy D.","family":"Lancaster","sequence":"additional","affiliation":[{"name":"Institute of Biophysics, Department of Structural Biology, Center of Human and Molecular Biology (ZHMB), Medical School, Saarland University, 66421 Homburg, Germany"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-1457-6222","authenticated-orcid":false,"given":"Frank","family":"Schmitz","sequence":"additional","affiliation":[{"name":"Institute of Anatomy and Cell Biology, Department of Neuroanatomy, Medical School, Saarland University, 66421 Homburg, Germany"}],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"1968","published-online":{"date-parts":[[2022,6,27]]},"reference":[{"key":"ref_1","doi-asserted-by":"crossref","first-page":"83","DOI":"10.1016\/j.ceb.2017.03.012","article-title":"Gates for soluble and membrane proteins, and two trafficking systems (IFT and LIFT), establish a dynamic ciliary signaling compartment","volume":"47","author":"Jensen","year":"2017","journal-title":"Curr. 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