Cyp1b1 Regulates Ocular Fissure Closure Through a Retinoic Acid-Independent Pathway.

ABSTRACT:

Purpose

Mutations in the CYP1B1 gene are the most commonly identified genetic causes of primary infantile-onset glaucoma. Despite this disease association, the role of CYP1B1 in eye development and its in vivo substrate remain unknown. In the present study, we used zebrafish to elucidate the mechanism by which cyp1b1 regulates eye development.

Methods

Zebrafish eye and neural crest development were analyzed using live imaging of transgenic zebrafish embryos, in situ hybridization, immunostaining, TUNEL assay, and methylacrylate sections. Cyp1b1 and retinoic acid (RA) levels were genetically (morpholino oligonucleotide antisense and mRNA) and pharmacologically manipulated to examine gene function.

Results

Using zebrafish, we observed that cyp1b1 was expressed in a spec

SUBMITTER: Williams AL 

PROVIDER: S-EPMC5308778 | biostudies-literature | 2017 Feb

REPOSITORIES: biostudies-literature