Optic cup and facial patterning defects in ocular ectoderm β-catenin gain-of-function mice

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Optic cup and facial patterning defects in ocular ectoderm β-catenin gain-of-function mice

Publisher
BioMed Central
Publication Date
Mar 15, 2006
Source
PMC
Keywords
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Abstract

1471-213X-6-14.fm ral ss BioMed CentBMC Developmental Biology Open AcceResearch article Optic cup and facial patterning defects in ocular ectoderm β-catenin gain-of-function mice Leigh-Anne D Miller1, April N Smith1, M Mark Taketo2 and Richard A Lang*1 Address: 1Division of Developmental Biology, Department of Ophthalmology, Children's Hospital Research Foundation and The University of Cincinnati, Cincinnati, OH 45229-3039, USA and 2Department of Pharmacology, Kyoto University School of Medicine, Kyoto, Japan Email: Leigh-Anne D Miller - [email protected]; April N Smith - [email protected]; M Mark Taketo - [email protected] tokyo.ac.jp; Richard A Lang* - [email protected] * Corresponding author Abstract Background: The canonical Wnt signaling pathway has a number of critical functions during embryonic development and, when activated aberrantly, in the genesis of cancer. Current evidence suggests that during eye development, regulation of Wnt signaling is critical for patterning the surface ectoderm that will contribute to multiple components of the eye. Wnt signaling loss-of- function experiments show that a region of periocular ectoderm will form ectopic lentoid bodies unless the Wnt pathway modifies its fate towards other structures. Consistent with this, Wnt signaling gain of function in the ocular region ectoderm results in a suppression of lens fate. Results: Here we demonstrate that ectoderm-specific Wnt signaling gain-of-function embryos exhibit additional defects besides those noted in the lens. There are profound facial defects including a foreshortened snout, malformation of the nasal region, and clefting of the epidermis along the ocular-nasal axis. Furthermore, despite the restriction of Wnt pathway gain-of-function to the surface ectoderm, the optic cup is inappropriately patterned and ultimately forms a highly convoluted, disorganized array of epithelium with the characteristics of retina and retinal pigmented epithelium. Conclusion: We

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