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
  • Zebrafish Proteins*/genetics
  • Zebrafish Proteins*/metabolism
  • Zebrafish*/genetics
  • Cell Survival/genetics
  • Eye
  • Morphogenesis/genetics
  • Mesoderm/metabolism
  • Animals
  • PAX2 Transcription Factor/genetics
  • PAX2 Transcription Factor/metabolism
(all 10)
PubMed
34535934 Full text @ Dev. Dyn.
Abstract
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.
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.
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
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Expression
Phenotype
Mutations / Transgenics
Allele Construct Type Affected Genomic Region
ba4TgTransgenic Insertion
    s896TgTransgenic Insertion
      sa10953
        Point Mutation
        tu29a
          Point Mutation
          z200TgTransgenic Insertion
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            Human Disease / Model
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            Fish
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            Engineered Foreign Genes
            Marker Marker Type Name
            EGFPEFGEGFP
            GFPEFGGFP
            mCherryEFGmCherry
            1 - 3 of 3
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