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- W2519409874 abstract "A critical aspect of vertebrate eye development is closure of the choroid fissure (CF). Defects in CF closure result in colobomas, which are a significant cause of childhood blindness worldwide. Despite the growing number of mutated loci associated with colobomas, we have a limited understanding of the cell biological underpinnings of CF closure. Here, we utilize the zebrafish embryo to identify key phases of CF closure and regulators of the process. Utilizing Laminin-111 as a marker for the basement membrane (BM) lining the CF, we determine the spatial and temporal patterns of BM breakdown in the CF, a prerequisite for CF closure. Similarly, utilizing a combination of in vivo time-lapse imaging, β-catenin immunohistochemistry and F-actin staining, we determine that tissue fusion, which serves to close the fissure, follows BM breakdown closely. Periocular mesenchyme (POM)-derived endothelial cells, which migrate through the CF to give rise to the hyaloid vasculature, possess distinct actin foci that correlate with regions of BM breakdown. Disruption of talin1, which encodes a regulator of the actin cytoskeleton, results in colobomas and these correlate with structural defects in the hyaloid vasculature and defects in BM breakdown. cloche mutants, which entirely lack a hyaloid vasculature, also possess defects in BM breakdown in the CF. Taken together, these data support a model in which the hyaloid vasculature and/or the POM-derived endothelial cells that give rise to the hyaloid vasculature contribute to BM breakdown during CF closure." @default.
- W2519409874 created "2016-09-23" @default.
- W2519409874 creator A5010834737 @default.
- W2519409874 creator A5011303424 @default.
- W2519409874 creator A5022824868 @default.
- W2519409874 creator A5023221767 @default.
- W2519409874 creator A5045032584 @default.
- W2519409874 creator A5052771197 @default.
- W2519409874 date "2016-11-01" @default.
- W2519409874 modified "2023-10-14" @default.
- W2519409874 title "The hyaloid vasculature facilitates basement membrane breakdown during choroid fissure closure in the zebrafish eye" @default.
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