ZFIN ID: ZDB-PUB-140710-9
aldh7a1 Regulates Eye and Limb Development in Zebrafish
Babcock, H.E., Dutta, S., Alur, R.P., Brocker, C., Vasiliou, V., Vitale, S., Abu-Asab, M., Brooks, B.P.
Date: 2014
Source: PLoS One   9: e101782 (Journal)
Registered Authors: Brooks, Brian P., Dutta, Sunit, Vasiliou, Vasilis
Keywords: none
MeSH Terms:
  • Aldehyde Dehydrogenase/genetics*
  • Animals
  • Cell Proliferation
  • DNA-Binding Proteins/genetics
  • Embryo, Nonmammalian
  • Embryonic Development/genetics
  • Extremities/embryology*
  • Eye/embryology
  • Eye/metabolism*
  • Gene Expression
  • Gene Expression Regulation, Developmental
  • Gene Knockdown Techniques
  • Phenotype
  • Tretinoin/pharmacology
  • Zebrafish/embryology*
  • Zebrafish/genetics*
  • Zebrafish Proteins/genetics
PubMed: 25004007 Full text @ PLoS One
Uveal coloboma is a potentially blinding congenital ocular malformation caused by failure of the optic fissure to close during development. Although mutations in numerous genes have been described, these account for a minority of cases, complicating molecular diagnosis and genetic counseling. Here we describe a key role of aldh7a1 as a gene necessary for normal eye development. We show that morpholino knockdown of aldh7a1 in zebrafish causes uveal coloboma and misregulation of nlz1, another known contributor to the coloboma phenotype, as well as skeletal abnormalities. Knockdown of aldh7a1 leads to reduced cell proliferation in the optic cup of zebrafish, delaying the approximation of the edges of the optic fissure. The aldh7a1 morphant phenotype is partially rescued by co-injection of nlz1 mRNA suggesting that nlz1 is functionally downstream of aldh7a1 in regulating cell proliferation in the optic cup. These results support a role of aldh7a1 in ocular development and skeletal abnormalities in zebrafish.