Abnormal vasculature interferes with optic fissure closure in lmo2 mutant zebrafish embryos

Omri Weiss, Rivka Kaufman, Natali Michaeli, Adi Inbal

Research output: Contribution to journalArticlepeer-review

Abstract

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.

Original languageAmerican English
Pages (from-to)191-198
Number of pages8
JournalDevelopmental Biology
Volume369
Issue number2
DOIs
StatePublished - 15 Sep 2012

Keywords

  • Coloboma
  • Lmo2
  • Ocular vasculature
  • Optic fissure
  • Zebrafish

All Science Journal Classification (ASJC) codes

  • Molecular Biology
  • Developmental Biology
  • Cell Biology

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