PUBLICATION

Abnormal retinal development in cloche mutant zebrafish

Authors
Dhakal, S., Stevens, C.B., Sebbagh, M., Weiss, O., Frey, R.A., Adamson, S., Shelden, E.A., Inbal, A., Stenkamp, D.L.
ID
ZDB-PUB-150819-5
Date
2015
Source
Developmental Dynamics : an official publication of the American Association of Anatomists   244(11): 1439-55 (Journal)
Registered Authors
Keywords
Müller glia, Retina, neurod1, neurogenesis, pax6a, photoreceptors, vasculature, zebrafish
MeSH Terms
  • Neurons/physiology
  • Hypoxia
  • Cell Differentiation
  • Zebrafish Proteins/genetics*
  • Zebrafish Proteins/physiology
  • Embryo, Nonmammalian/metabolism
  • Mutation*
  • Phenotype
  • Neuroglia/physiology
  • Cell Survival
  • Retina/abnormalities*
  • Retina/embryology*
  • Basic Helix-Loop-Helix Transcription Factors/genetics*
  • Basic Helix-Loop-Helix Transcription Factors/physiology
  • Microscopy, Confocal
  • Animals
  • Cell Proliferation
  • Zebrafish/genetics*
  • Cell Death
  • Animals, Genetically Modified
  • Retinal Pigment Epithelium/metabolism
  • Stem Cells
  • Gene Expression Regulation, Developmental
(all 23)
PubMed
26283463 Full text @ Dev. Dyn.
Abstract
Background Functions for the early embryonic vasculature in regulating development of central nervous system tissues, such as the retina, have been suggested by in vitro studies and by in vivo manipulations that caused additional ocular vessels to develop. Here we use an avascular zebrafish embryo, cloche-/- (clo-/-), to begin to identify necessary developmental functions of the ocular vasculature in regulating development and patterning of the neural retina, in vivo. These studies are possible in zebrafish embryos, which do not yet rely upon the vasculature for tissue oxygenation.
Results clo-/- embryos lacked early ocular vasculature and were microphthalmic, with reduced retinal cell proliferation and cell survival. Retinas of clo mutants were disorganized, with irregular synaptic layers, mispatterned expression domains of retinal transcription factors, morphologically abnormal Müller glia, reduced differentiation of specific retinal cell types, and sporadically distributed cone photoreceptors. Blockade of p53-mediated cell death did not completely rescue this phenotype and revealed ectopic cones in the inner nuclear layer. clo-/- embryos did not upregulate a molecular marker for hypoxia.
Conclusions The disorganized retinal phenotype of clo-/- embryos is consistent with a neural and glial developmental patterning role for the early ocular vasculature that is independent of its eventual function in gas exchange. This article is protected by copyright. All rights reserved.
Genes / Markers
Figures
Figure Gallery (11 images) / 2
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Expression
No data available
Phenotype
No data available
Mutations / Transgenics
Allele Construct Type Affected Genomic Region
m39
    		Deficiency
    m378
      		Unknown
      s843TgTransgenic Insertion
        1 - 3 of 3
        Show
        Human Disease / Model
        No data available
        Sequence Targeting Reagents
        Target Reagent Reagent Type
        tp53MO4-tp53MRPHLNO
        1 - 1 of 1
        Show
        Fish
        No data available
        Antibodies
        Orthology
        No data available
        Engineered Foreign Genes
        Marker Marker Type Name
        EGFPEFGEGFP
        1 - 1 of 1
        Show
        Mapping
        No data available
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        Citation
        Dhakal, S., Stevens, C.B., Sebbagh, M., Weiss, O., Frey, R.A., Adamson, S., Shelden, E.A., Inbal, A., Stenkamp, D.L. (2015) Abnormal retinal development in cloche mutant zebrafish. Developmental Dynamics : an official publication of the American Association of Anatomists. 244(11):1439-55.