{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,10,2]],"date-time":"2026-10-02T22:21:07Z","timestamp":1790979667399,"version":"4.1.0"},"reference-count":38,"publisher":"National Academy of Sciences","issue":"36","content-domain":{"domain":["www.pnas.org"],"crossmark-restriction":true},"short-container-title":["Proc. Natl. Acad. Sci. U.S.A."],"published-print":{"date-parts":[[2006,9,5]]},"abstract":"<jats:p>\n                    Congenital hypertrophy\/hyperplasia of the retinal pigmented epithelium is an ocular lesion found in patients harboring mutations in the\n                    <jats:italic>adenomatous polyposis coli<\/jats:italic>\n                    (\n                    <jats:italic>APC<\/jats:italic>\n                    ) tumor suppressor gene. We report that Apc-deficient zebrafish display developmental abnormalities of both the lens and retina. Injection of dominant-negative Lef reduced Wnt signaling in the lens but did not rescue retinal differentiation defects. In contrast, treatment of\n                    <jats:italic>apc<\/jats:italic>\n                    mutants with all-trans retinoic acid rescued retinal differentiation defects but had no apparent effect on the lens. We identified Rdh5 as a retina-specific retinol dehydrogenase controlled by APC. Morpholino knockdown of Rdh5 phenocopied the\n                    <jats:italic>apc<\/jats:italic>\n                    mutant retinal differentiation defects and was rescued by treatment with exogenous all-trans retinoic acid. Microarray analyses of\n                    <jats:italic>apc<\/jats:italic>\n                    mutants and Rdh5 morphants revealed a profound overlap in the transcriptional profile of these embryos. These findings support a model wherein Apc serves a dual role in regulating Wnt and retinoic acid signaling within the eye and suggest retinoic acid deficiency as an explanation for\n                    <jats:italic>APC<\/jats:italic>\n                    mutation-associated retinal defects such as congenital hypertrophy\/hyperplasia of the retinal pigmented epithelium.\n                  <\/jats:p>","DOI":"10.1073\/pnas.0601634103","type":"journal-article","created":{"date-parts":[[2006,8,28]],"date-time":"2006-08-28T22:10:40Z","timestamp":1156803040000},"page":"13409-13414","update-policy":"https:\/\/doi.org\/10.1073\/pnas.cm10313","source":"Crossref","is-referenced-by-count":37,"title":["Dual roles for\n                    <i>adenomatous polyposis coli<\/i>\n                    in regulating retinoic acid biosynthesis and Wnt during ocular development"],"prefix":"10.1073","volume":"103","author":[{"given":"Lincoln D.","family":"Nadauld","sequence":"first","affiliation":[{"name":"Departments of *Oncological Sciences and"},{"name":"Huntsman Cancer Institute, University of Utah, Salt Lake City, UT 84112"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Stephanie","family":"Chidester","sequence":"additional","affiliation":[{"name":"Huntsman Cancer Institute, University of Utah, Salt Lake City, UT 84112"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Dawne N.","family":"Shelton","sequence":"additional","affiliation":[{"name":"Departments of *Oncological Sciences and"},{"name":"Huntsman Cancer Institute, University of Utah, Salt Lake City, UT 84112"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Kunal","family":"Rai","sequence":"additional","affiliation":[{"name":"Departments of *Oncological Sciences and"},{"name":"Huntsman Cancer Institute, University of Utah, Salt Lake City, UT 84112"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Talmage","family":"Broadbent","sequence":"additional","affiliation":[{"name":"Departments of *Oncological Sciences and"},{"name":"Huntsman Cancer Institute, University of Utah, Salt Lake City, UT 84112"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Imelda T.","family":"Sandoval","sequence":"additional","affiliation":[{"name":"Medicinal Chemistry and"},{"name":"Huntsman Cancer Institute, University of Utah, Salt Lake City, UT 84112"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Peter W.","family":"Peterson","sequence":"additional","affiliation":[{"name":"Huntsman Cancer Institute, University of Utah, Salt Lake City, UT 84112"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Elizabeth J.","family":"Manos","sequence":"additional","affiliation":[{"name":"Huntsman Cancer Institute, University of Utah, Salt Lake City, UT 84112"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Chris M.","family":"Ireland","sequence":"additional","affiliation":[{"name":"Medicinal Chemistry and"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"H. 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