PUBLICATION

Expression profiling during ocular development identifies 2 Nlz genes with a critical role in optic fissure closure

Authors
Brown, J.D., Dutta, S., Bharti, K., Bonner, R.F., Munson, P.J., Dawid, I.B., Akhtar, A.L., Onojafe, I.F., Alur, R.P., Gross, J.M., Hejtmancik, J.F., Jiao, X., Chan, W.Y., and Brooks, B.P.
ID
ZDB-PUB-090204-20
Date
2009
Source
Proceedings of the National Academy of Sciences of the United States of America   106(5): 1462-1467 (Journal)
Registered Authors
Brooks, Brian P., Dawid, Igor B., Dutta, Sunit, Gross, Jeffrey
Keywords
coloboma, eye, pax2, zinc finger protein, zebrafish
MeSH Terms
  • Animals
  • Base Sequence
  • Coloboma/genetics
  • DNA Primers
  • DNA-Binding Proteins/genetics*
  • Eye/embryology*
  • Gene Expression Profiling*
  • Gene Expression Regulation, Developmental
  • In Situ Hybridization
  • PAX2 Transcription Factor/genetics
  • Polymerase Chain Reaction
  • Promoter Regions, Genetic
  • RNA, Messenger/genetics
  • Repressor Proteins/genetics*
  • Zebrafish
  • Zebrafish Proteins/genetics*
  • Zinc Fingers
PubMed
19171890 Full text @ Proc. Natl. Acad. Sci. USA
Abstract
The gene networks underlying closure of the optic fissure during vertebrate eye development are poorly understood. Here, we profile global gene expression during optic fissure closure using laser capture microdissected (LCM) tissue from the margins of the fissure. From these data, we identify a unique role for the C(2)H(2) zinc finger proteins Nlz1 and Nlz2 in normal fissure closure. Gene knockdown of nlz1 and/or nlz2 in zebrafish leads to a failure of the optic fissure to close, a phenotype which closely resembles that seen in human uveal coloboma. We also identify misregulation of pax2 in the developing eye of morphant fish, suggesting that Nlz1 and Nlz2 act upstream of the Pax2 pathway in directing proper closure of the optic fissure.
Genes / Markers
Figures
Expression
Phenotype
Mutation and Transgenics
Human Disease / Model Data
Sequence Targeting Reagents
Fish
Antibodies
Orthology
Engineered Foreign Genes
Mapping
Errata and Notes