PUBLICATION
Pax2a, but not pax2b, influences cell survival and periocular mesenchyme localization to facilitate zebrafish optic fissure closure
- Authors
- Lusk, S., Kwan, K.M.
- ID
- ZDB-PUB-210919-2
- Date
- 2021
- Source
- Developmental Dynamics : an official publication of the American Association of Anatomists 251(4): 625-644 (Journal)
- Registered Authors
- Kwan, Kristen
- Keywords
- coloboma, optic cup, optic fissure, pax2a, pax2b, zebrafish
- MeSH Terms
-
- Animals
- Cell Survival/genetics
- Eye
- Mesoderm/metabolism
- Morphogenesis/genetics
- PAX2 Transcription Factor/genetics
- PAX2 Transcription Factor/metabolism
- Zebrafish*/genetics
- Zebrafish Proteins*/genetics
- Zebrafish Proteins*/metabolism
- PubMed
- 34535934 Full text @ Dev. Dyn.
Citation
Lusk, S., Kwan, K.M. (2021) Pax2a, but not pax2b, influences cell survival and periocular mesenchyme localization to facilitate zebrafish optic fissure closure. Developmental Dynamics : an official publication of the American Association of Anatomists. 251(4):625-644.
Abstract
Background Pax2 is required for optic fissure development in many organisms, including humans and zebrafish. Zebrafish loss-of-function mutations in pax2a display coloboma, yet the etiology of the morphogenetic defects is unclear. Further, pax2 is duplicated in zebrafish, and a role for pax2b in optic fissure development has not been examined.
Results Using a combination of imaging and molecular genetics, we interrogated a potential role for pax2b and examined how loss of pax2 affects optic fissure development. Although optic fissure formation appears normal in pax2 mutants, an endothelial-specific subset of periocular mesenchyme fails to initially localize within the optic fissure, yet both neural crest and endothelial-derived periocular mesenchyme ectopically accumulate at later stages in pax2a and pax2a; pax2b mutants. Apoptosis is not upregulated within the optic fissure in pax2 mutants, yet cell death is increased in tissues outside of the optic fissure, and when apoptosis is inhibited, coloboma is partially rescued. In contrast to pax2a, loss of pax2b does not appear to affect optic fissure morphogenesis.
Conclusions Our results suggest that pax2a, but not pax2b, supports cell survival outside of the optic fissure and periocular mesenchyme abundance within it to facilitate optic fissure closure. This article is protected by copyright. All rights reserved.
Genes / Markers
Expression
Phenotype
Mutations / Transgenics
Human Disease / Model
Sequence Targeting Reagents
Fish
Orthology
Engineered Foreign Genes
Mapping