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• W2912135638 endingPage "72" @default.
• W2912135638 startingPage "66" @default.
• W2912135638 abstract "Mutation of FOXC1 causes Axenfeld-Rieger Syndrome (ARS) with early onset or congenital glaucoma. We assessed retinal ganglion cell (RGC) number in zebrafish due to CRISPR-mediated mutation and antisense inhibition of two-forkhead box transcription factors, foxc1a and foxc1b. These genes represent duplicated homologues of human FOXC1. Using a CRISPR induced null mutation in foxc1b, in combination with antisense inhibition of foxc1a, we demonstrate reduced cell number in the retinal ganglion cell layer of developing zebrafish eyes. As early as 5 days post fertilization (dpf), fewer RGCs are found in foxc1b homozygous mutants injected with foxc1a morpholinos, and a thinner optic nerve results. Our data illustrates that foxc1 is required for the expression of atonal homolog 7 (atoh7), a gene that is necessary for RGC differentiation. As markers of differentiated RGCs (pou4f2) are downregulated in foxc1b-/- mutants injected with foxc1a morpholinos and no cell death is observed, our results are consistent with defects in the differentiation of RGCs leading to reduced cell number, as opposed to increased cell death of RGCs or off targets effects of morpholino injection. Our zebrafish model demonstrates that aberrant regulation of RGC number could act in concert with other known glaucoma risk factors to influence the development of congenital and early onset glaucoma due to FOXC1 mutation." @default.
• W2912135638 created "2019-02-21" @default.
• W2912135638 creator A5003960852 @default.
• W2912135638 creator A5010791496 @default.
• W2912135638 creator A5015361948 @default.
• W2912135638 date "2019-03-01" @default.
• W2912135638 modified "2023-10-16" @default.
• W2912135638 title "Loss of foxc1 in zebrafish reduces optic nerve size and cell number in the retinal ganglion cell layer" @default.
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• W2912135638 doi "https://doi.org/10.1016/j.visres.2019.01.008" @default.
• W2912135638 hasPubMedId "https://pubmed.ncbi.nlm.nih.gov/30684501" @default.
• W2912135638 hasPublicationYear "2019" @default.
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