{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,5,18]],"date-time":"2026-05-18T16:13:12Z","timestamp":1779120792835,"version":"3.51.4"},"reference-count":58,"publisher":"Wiley","issue":"4","license":[{"start":{"date-parts":[[2006,6,6]],"date-time":"2006-06-06T00:00:00Z","timestamp":1149552000000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/onlinelibrary.wiley.com\/termsAndConditions#vor"}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":["Pigment Cell Research"],"published-print":{"date-parts":[[2006,8]]},"abstract":"<jats:title>Summary<\/jats:title><jats:p>Hypopigmentation is a characteristic of several diseases associated with vesicle traffic defects, like the Hermansky\u2013Pudlak, Chediak\u2013Higashi, and Griscelli syndromes. Hypopigmentation is also a characteristic of the zebrafish mutant <jats:italic>vps18<\/jats:italic><jats:sup><jats:italic>hi2499A<\/jats:italic><\/jats:sup>, which is affected in the gene <jats:italic>vps18<\/jats:italic>, a component of the homotypic fusion and protein sorting complex that is involved in tethering during vesicular traffic. Vps18, as part of this complex, participates in the formation of early endosomes, late endosomes, and lysosomes. Here, we show that Vps18 is also involved in the formation of melanosomes. In the zebrafish mutant <jats:italic>vps18<\/jats:italic><jats:sup><jats:italic>hi2499A<\/jats:italic><\/jats:sup> the retroviral insertion located at exon 4 of <jats:italic>vps18<\/jats:italic>, leads to the formation of two abnormal splicing variants lacking the coding sequence for the clathrin repeat and the RING finger conserved domains. A deficiency of Vps18 in zebrafish larvae results in hepatomegaly and skin hypopigmentation. We also observed a drastic reduction in the number of melanosomes in the eye's retinal pigmented epithelium along with the accumulation of immature melanosomes. A significant reduction in the <jats:italic>vps18<\/jats:italic><jats:sup><jats:italic>hi2499A<\/jats:italic><\/jats:sup> larvae visual system capacity was found using the optokinetic response assay. We propose that the insertional mutant <jats:italic>vps18<\/jats:italic><jats:sup><jats:italic>hi2499A<\/jats:italic><\/jats:sup> can be used as a model for studying hypopigmentation diseases in which vesicle traffic problems exist.<\/jats:p>","DOI":"10.1111\/j.1600-0749.2006.00320.x","type":"journal-article","created":{"date-parts":[[2006,6,7]],"date-time":"2006-06-07T01:32:39Z","timestamp":1149643959000},"page":"315-326","source":"Crossref","is-referenced-by-count":43,"title":["The zebrafish mutant <i>vps18<\/i> as a model for vesicle\u2010traffic related hypopigmentation diseases"],"prefix":"10.1111","volume":"19","author":[{"given":"Ernesto","family":"Maldonado","sequence":"first","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Fabiola","family":"Hernandez","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Carlos","family":"Lozano","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Marta E.","family":"Castro","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Rosa E.","family":"Navarro","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"311","published-online":{"date-parts":[[2006,6,6]]},"reference":[{"key":"e_1_2_6_2_1","doi-asserted-by":"publisher","DOI":"10.1006\/dbio.1999.9462"},{"key":"e_1_2_6_3_1","doi-asserted-by":"publisher","DOI":"10.1016\/S0091-679X(04)77001-6"},{"key":"e_1_2_6_4_1","doi-asserted-by":"publisher","DOI":"10.1073\/pnas.0403929101"},{"key":"e_1_2_6_5_1","doi-asserted-by":"publisher","DOI":"10.1371\/journal.pbio.0020139"},{"key":"e_1_2_6_6_1","doi-asserted-by":"publisher","DOI":"10.1152\/jn.00311.2004"},{"key":"e_1_2_6_7_1","doi-asserted-by":"publisher","DOI":"10.1016\/S0092-8674(03)01079-1"},{"key":"e_1_2_6_8_1","doi-asserted-by":"publisher","DOI":"10.1073\/pnas.92.23.10545"},{"key":"e_1_2_6_9_1","doi-asserted-by":"publisher","DOI":"10.1242\/dev.128.12.2385"},{"key":"e_1_2_6_10_1","doi-asserted-by":"publisher","DOI":"10.1128\/jvi.76.5.2192-2198.2002"},{"key":"e_1_2_6_11_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.devcel.2004.05.002"},{"key":"e_1_2_6_12_1","doi-asserted-by":"crossref","first-page":"593","DOI":"10.1007\/s00427-002-0279-3","article-title":"Phenylthiourea disrupts thyroid function in developing zebrafish","volume":"212","author":"Elsalini O.A.","year":"2003","journal-title":"Dev. 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