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Nadauld, LD, Chidester, S, Shelton, DN, Rai, K, Broadbent, T, Sandoval, IT, Peterson, PW, Manos, EJ, Ireland, CM, Yost, HJ and Jones, DA (2006) Dual roles for adenomatous polyposis coli in regulating retinoic acid biosynthesis and Wnt during ocular development. Proc. Natl. Acad. Sci. U.S.A. 103:13409-14


Congenital hypertrophy/hyperplasia of the retinal pigmented epithelium is an ocular lesion found in patients harboring mutations in the adenomatous polyposis coli (APC) 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 apc 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 apc mutant retinal differentiation defects and was rescued by treatment with exogenous all-trans retinoic acid. Microarray analyses of apc 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 APC mutation-associated retinal defects such as congenital hypertrophy/hyperplasia of the retinal pigmented epithelium.


PubMed PMC1569177 Online version:10.1073/pnas.0601634103


Adenomatous Polyposis Coli Protein/deficiency; Adenomatous Polyposis Coli Protein/genetics; Adenomatous Polyposis Coli Protein/metabolism; Animals; Eye/drug effects; Eye/embryology; Gene Expression Regulation, Developmental; Homozygote; Microinjections; Molecular Sequence Data; Mutation; Oligonucleotides, Antisense/pharmacology; Tretinoin/metabolism; Tretinoin/pharmacology; Wnt Proteins/metabolism; Zebrafish/embryology; Zebrafish/genetics



Gene product Qualifier GO ID GO term name Evidence Code with/from Aspect Notes Status

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