Abnormal vasculature interferes with optic fissure closure in lmo2 mutant zebrafish embryos
- Authors
- Weiss, O., Kaufman, R., Michaeli, N., and Inbal, A.
- ID
- ZDB-PUB-120724-24
- Date
- 2012
- Source
- Developmental Biology 369(2): 191-198 (Journal)
- Registered Authors
- Inbal, Adi
- Keywords
- zebrafish, optic fissure, coloboma, ocular vasculature, lmo2
- MeSH Terms
-
- Zebrafish Proteins/genetics*
- Zebrafish/embryology*
- Zebrafish/genetics*
- Phenotype
- Gene Expression Regulation, Developmental
- Coloboma/embryology
- Coloboma/genetics
- Mutation
- Animals
- Transcription Factors/genetics*
- Eye/blood supply
- Eye/enzymology
- Retinal Vessels/abnormalities
- Retinal Vessels/embryology
- Eye Abnormalities/embryology*
- Eye Abnormalities/genetics*
- Animals, Genetically Modified
- LIM Domain Proteins/genetics*
- Base Sequence
- DNA Primers/genetics
- PubMed
- 22819672 Full text @ Dev. Biol.
Ocular coloboma is a potentially blinding congenital eye malformation caused by failure of optic fissure closure during early embryogenesis. The optic fissure is a ventral groove that forms during optic cup morphogenesis, and through which hyaloid artery and vein enter and leave the developing eye, respectively. After hyaloid artery and vein formation, the optic fissure closes around them. The mechanisms underlying optic fissure closure are poorly understood, and whether and how this process is influenced by hyaloid vessel development is unknown. Here we show that a loss-of-function mutation in lmo2, a gene specifically required for hematopoiesis and vascular development, results in failure of optic fissure closure in zebrafish. Analysis of ocular blood vessels in lmo2 mutants reveals that some vessels are severely dilated, including the hyaloid vein. Remarkably, reducing vessel size leads to rescue of optic fissure phenotype. Our results reveal a new mechanism leading to coloboma, whereby malformed blood vessels interfere with eye morphogenesis.