April 2010
Volume 51, Issue 13
Free
ARVO Annual Meeting Abstract  |   April 2010
The Zebrafish Patched2 Mutant as a Model for the Study of Basal Cell Naevus Syndrome (BCNS)-Related Ocular Pathologies
Author Affiliations & Notes
  • J. Bibliowicz
    Molecular Cell and Developmental Biology, The University of Texas at Austin, Austin, Texas
  • J. M. Gross
    Molecular Cell and Developmental Biology, The University of Texas at Austin, Austin, Texas
  • Footnotes
    Commercial Relationships  J. Bibliowicz, None; J.M. Gross, None.
  • Footnotes
    Support  NIH RO1 EY18005
Investigative Ophthalmology & Visual Science April 2010, Vol.51, 4028. doi:
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    • Get Citation

      J. Bibliowicz, J. M. Gross; The Zebrafish Patched2 Mutant as a Model for the Study of Basal Cell Naevus Syndrome (BCNS)-Related Ocular Pathologies. Invest. Ophthalmol. Vis. Sci. 2010;51(13):4028.

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      © ARVO (1962-2015); The Authors (2016-present)

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Abstract

Purpose: : Müller glia are retina-specific glia that normally maintain neuronal health and retinal organization, and are known to become ‘reactive’ upon exposure to growth factors or in response to retinal injury. Our previous characterization of the embryonic zebrafish patched2 mutant retina revealed localized upregulation of GFAP, a marker for 'reactive' glia, as well as morphological abnormalities at the vitreo-retinal interface, where Müller glial endfeet terminate. Here, we extend our analysis of the patched2 mutant retina to later developmental stages to investigate whether Müller glia homeostasis and retinal organization are perturbed when Hedgehog pathway activity is upregulated.

Methods: : Histological and immunohistochemical analyses were utilized to analyze retinal architecture and Müller glial reactivity in juvenile patched2 mutants and their heterozygous and homozygous siblings.

Results: : Analysis of the juvenile patched2 mutant retina revealed upregulation of GFAP, a marker for 'reactive' glia, throughout the central retina. Ectopic proliferation and rosette formation were detected in the central retina and these abnormalities were spatially associated with displaced Müller glia. Current research efforts are focused on determining whether Müller glia reactivity may play a role in the onset of these retinal abnormalities, and/or whether Müller glia are displaced in response to these defects.

Conclusions: : patched2

Keywords: retinal development • Muller cells • retinal glia 
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