Optic cup and facial patterning defects in ocular ectoderm β-catenin gain-of-function mice
| Autor: | Makoto Mark Taketo, Leigh Anne D. Miller, April N. Smith, Richard A. Lang |
|---|---|
| Jazyk: | angličtina |
| Rok vydání: | 2006 |
| Předmět: |
Beta-catenin
Time Factors Ectoderm Mice Transgenic Mice Lens Crystalline medicine Animals Transgenes lcsh:QH301-705.5 beta Catenin Body Patterning biology Embryogenesis Wnt signaling pathway Gene Expression Regulation Developmental Anatomy Embryo Mammalian Surface ectoderm eye diseases Cell biology Wnt Proteins medicine.anatomical_structure lcsh:Biology (General) Catenin Lens (anatomy) Face Mutation biology.protein Eye development sense organs Developmental Biology Research Article Signal Transduction |
| Zdroj: | BMC Developmental Biology, Vol 6, Iss 1, p 14 (2006) BMC Developmental Biology |
| Popis: | 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 suggest that activation of the Wnt pathway in surface ectoderm may disrupt the normal exchange of signals between the presumptive lens and retina that coordinate development of a functional eye. |
| Databáze: | OpenAIRE |
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