Browsing by Subject "Lens"
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Item Integrin alphα5/fibronectin1 and focal adhesion kinase are required for lens fiber morphogenesis in zebrafish(2010-08) Hayes, Julie Marie; Gross, Jeffrey Martin; Wallingford, John B.Fibronectin (fn) and integrin α5 (itgα5) are both key players in cell adhesion and intracellar signaling, however the specific in vivo role of these proteins has never been analyzed in the vertebrate lens. The results presented here indicate that Fn1 and Itgα5 proteins are essential for the proper development of the lens. The loss of Fn1 protein in the zebrafish embryo results in distinct adhesion defects, defects in lens fiber morphogenesis, and cataracts. These results were phenocopied in zebrafish itga5 mutants, thereby indicating an essential role for Fn1 and Itgα5 during lens development. Furthermore, embryos with reduced levels of ptk2.1 (focal adhesion kinase – FAK) also phenocopied the defective fn1 and itgα5 lens, suggesting that FAK is a major player in the intracellular signaling mediated by Fn1/Itgα5 interactions in the lens.Item The roles of mab21l2 in development of the eye(2019-07-23) Gath, Natalie Nicole; Vokes, Steven Alexander; Gross, Jeffrey Martin; Agarwala, Seema; Matouschek, Andreas; Stein, DavidMutations in MAB21L2 result in severe ocular defects including microphthalmia, anophthalmia, coloboma, microcornea, and cataracts. The molecular and cellular underpinnings of these defects are unknown, as is the normal cellular function of MAB21L2. Zebrafish mab21l2 [superscript au10] mutants possess ocular defects resembling those in humans with MAB21L2 mutations, providing an excellent model to characterize mab21l2 functions during eye development. mab21l2 [superscript -/-] mutants possessed a host of ocular defects including microphthalmia and colobomas as well as small, disorganized lenses and cornea dysgenesis. Decreased proliferation, increased cell death, and defects in marker gene expression were detected in the lens. Cell death in the optic stalk was elevated in mab21l2 [superscript -/-] mutants and the basement membrane between the edges of the choroid fissure failed to break down. Neuronal differentiation in the retina was normal, however. mab21l2 [superscript -/-] mutant corneas were disorganized, possessed an increased number of cells, some of which proliferated ectopically, and failed to differentiate the corneal stroma. Human mutant MAB21L2 [superscript R51C] and MAB21L2 [superscript R51H] mRNAs possessed dominant negative function, inducing colobomas in wild type fish. Yeast-2-hybrid assays provided potential binding partners for the function of mab21l2, including transcription factors and actin/myosin related proteins. mab21l2 function is required for morphogenesis and cell survival in the lens and optic cup, and basement membrane breakdown in the choroid fissure. mab21l2 function also regulates proliferation in the lens and cornea; in its absence, the lens is small and mispatterned, and corneal morphogenesis and patterning are also disrupted. mab21l2 protein function may involve transcriptional regulation or control of cell shape and movement.