PUBLICATION
Loss of zebrafish dzip1 results in inappropriate recruitment of periocular mesenchyme to the optic fissure and ocular coloboma
- Authors
- Nandamuri, S.P., Lusk, S., Kwan, K.M.
- ID
- ZDB-PUB-220315-33
- Date
- 2022
- Source
- PLoS One 17: e0265327 (Journal)
- Registered Authors
- Kwan, Kristen
- Keywords
- none
- MeSH Terms
-
- Adaptor Proteins, Signal Transducing/metabolism
- Animals
- Carrier Proteins/metabolism*
- Coloboma*/genetics
- Eye/metabolism
- Mesoderm/metabolism
- Mice
- Optic Disk*/metabolism
- Zebrafish/genetics
- Zebrafish Proteins/genetics
- Zebrafish Proteins/metabolism
- PubMed
- 35286359 Full text @ PLoS One
Citation
Nandamuri, S.P., Lusk, S., Kwan, K.M. (2022) Loss of zebrafish dzip1 results in inappropriate recruitment of periocular mesenchyme to the optic fissure and ocular coloboma. PLoS One. 17:e0265327.
Abstract
Cilia are essential for the development and function of many different tissues. Although cilia machinery is crucial in the eye for photoreceptor development and function, a role for cilia in early eye development and morphogenesis is still somewhat unclear: many zebrafish cilia mutants retain cilia at early stages due to maternal deposition of cilia components. An eye phenotype has been described in the mouse Arl13 mutant, however, zebrafish arl13b is maternally deposited, and an early role for cilia proteins has not been tested in zebrafish eye development. Here we use the zebrafish dzip1 mutant, which exhibits a loss of cilia throughout stages of early eye development, to examine eye development and morphogenesis. We find that in dzip1 mutants, initial formation of the optic cup proceeds normally, however, the optic fissure subsequently fails to close and embryos develop the structural eye malformation ocular coloboma. Further, neural crest cells, which are implicated in optic fissure closure, do not populate the optic fissure correctly, suggesting that their inappropriate localization may be the underlying cause of coloboma. Overall, our results indicate a role for dzip1 in proper neural crest localization in the optic fissure and optic fissure closure.
Genes / Markers
Expression
Phenotype
Mutations / Transgenics
Human Disease / Model
Sequence Targeting Reagents
Fish
Orthology
Engineered Foreign Genes
Mapping