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
-
- Gene Expression Profiling*
- Base Sequence
- Coloboma/genetics
- Promoter Regions, Genetic
- Zebrafish
- PAX2 Transcription Factor/genetics
- RNA, Messenger/genetics
- Repressor Proteins/genetics*
- Animals
- Polymerase Chain Reaction
- DNA-Binding Proteins/genetics*
- In Situ Hybridization
- Gene Expression Regulation, Developmental
- Zebrafish Proteins/genetics*
- Zinc Fingers
- DNA Primers
- Eye/embryology*
- PubMed
- 19171890 Full text @ Proc. Natl. Acad. Sci. USA
Citation
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. (2009) Expression profiling during ocular development identifies 2 Nlz genes with a critical role in optic fissure closure. Proceedings of the National Academy of Sciences of the United States of America. 106(5):1462-1467.
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
Expression
Phenotype
Mutations / Transgenics
Human Disease / Model
Sequence Targeting Reagents
Fish
Orthology
Engineered Foreign Genes
Mapping