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Dev Dyn. 2015 Nov;244(11):1439-1455. doi: 10.1002/dvdy.24322. Epub 2015 Sep 2.

Abnormal retinal development in Cloche mutant zebrafish.

Author information

1
Department of Biological Sciences, University of Idaho, Moscow, ID USA.
2
Neuroscience Graduate Program, University of Idaho, Moscow, ID USA.
3
Department of Medical Neurobiology, Institute for Medical Research Israel-Canada, The Hebrew University-Hadassah Medical School, Jerusalem, Israel.
4
Department of Molecular Biosciences, Washington State University, Pullman, WA USA.
#
Contributed equally

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.

KEYWORDS:

Müller glia; neurod1; neurogenesis; pax6a; photoreceptors; retina; vasculature; zebrafish

PMID:
26283463
PMCID:
PMC4619121
DOI:
10.1002/dvdy.24322
[Indexed for MEDLINE]
Free PMC Article

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