Zfp503/Nlz2 Is Required for RPE Differentiation and Optic Fissure Closure.

ABSTRACT:

Purpose

Uveal coloboma is a congenital eye malformation caused by failure of the optic fissure to close in early human development. Despite significant progress in identifying genes whose regulation is important for executing this closure, mutations are detected in a minority of cases using known gene panels, implying additional genetic complexity. We have previously shown knockdown of znf503 (the ortholog of mouse Zfp503) in zebrafish causes coloboma. Here we characterize Zfp503 knockout (KO) mice and evaluate transcriptomic profiling of mutant versus wild-type (WT) retinal pigment epithelium (RPE)/choroid.

Methods

Zfp503 KO mice were generated by gene targeting using homologous recombination. Embryos were characterized grossly and histologically. Patterns and level of deve

SUBMITTER: Boobalan E 

PROVIDER: S-EPMC9645360 | biostudies-literature | 2022 Nov

REPOSITORIES: biostudies-literature