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Loss of Crb2b-lf leads to anterior segment defects in old zebrafish

Defects in the retina or the anterior segment of the eye lead to compromised vision and affect millions of people. Understanding how these ocular structures develop and are maintained is therefore of paramount importance. The maintenance of proper vision depends, among other factors, on the function...

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Autores principales: Kujawski, Satu, Crespo, Cátia, Luz, Marta, Yuan, Michaela, Winkler, Sylke, Knust, Elisabeth
Formato: Online Artículo Texto
Lenguaje:English
Publicado: The Company of Biologists Ltd 2020
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC7044448/
https://www.ncbi.nlm.nih.gov/pubmed/31988089
http://dx.doi.org/10.1242/bio.047555
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author Kujawski, Satu
Crespo, Cátia
Luz, Marta
Yuan, Michaela
Winkler, Sylke
Knust, Elisabeth
author_facet Kujawski, Satu
Crespo, Cátia
Luz, Marta
Yuan, Michaela
Winkler, Sylke
Knust, Elisabeth
author_sort Kujawski, Satu
collection PubMed
description Defects in the retina or the anterior segment of the eye lead to compromised vision and affect millions of people. Understanding how these ocular structures develop and are maintained is therefore of paramount importance. The maintenance of proper vision depends, among other factors, on the function of genes controlling apico-basal polarity. In fact, mutations in polarity genes are linked to retinal degeneration in several species, including human. Here we describe a novel zebrafish crb2b allele (crb2b(e40)), which specifically affects the crb2b long isoform. crb2b(e40) mutants are viable and display normal ocular development. However, old crb2b(e40) mutant fish develop multiple defects in structures of the anterior segment, which includes the cornea, the iris and the lens. Phenotypes are characterised by smaller pupils due to expansion of the iris and tissues of the iridocorneal angle, an increased number of corneal stromal keratocytes, an abnormal corneal endothelium and an expanded lens capsule. These findings illustrate a novel role for crb2b in the maintenance of the anterior segment and hence add an important function to this polarity regulator, which may be conserved in other vertebrates including humans.
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spelling pubmed-70444482020-02-27 Loss of Crb2b-lf leads to anterior segment defects in old zebrafish Kujawski, Satu Crespo, Cátia Luz, Marta Yuan, Michaela Winkler, Sylke Knust, Elisabeth Biol Open Research Article Defects in the retina or the anterior segment of the eye lead to compromised vision and affect millions of people. Understanding how these ocular structures develop and are maintained is therefore of paramount importance. The maintenance of proper vision depends, among other factors, on the function of genes controlling apico-basal polarity. In fact, mutations in polarity genes are linked to retinal degeneration in several species, including human. Here we describe a novel zebrafish crb2b allele (crb2b(e40)), which specifically affects the crb2b long isoform. crb2b(e40) mutants are viable and display normal ocular development. However, old crb2b(e40) mutant fish develop multiple defects in structures of the anterior segment, which includes the cornea, the iris and the lens. Phenotypes are characterised by smaller pupils due to expansion of the iris and tissues of the iridocorneal angle, an increased number of corneal stromal keratocytes, an abnormal corneal endothelium and an expanded lens capsule. These findings illustrate a novel role for crb2b in the maintenance of the anterior segment and hence add an important function to this polarity regulator, which may be conserved in other vertebrates including humans. The Company of Biologists Ltd 2020-02-11 /pmc/articles/PMC7044448/ /pubmed/31988089 http://dx.doi.org/10.1242/bio.047555 Text en © 2020. Published by The Company of Biologists Ltd http://creativecommons.org/licenses/by/4.0This is an Open Access article distributed under the terms of the Creative Commons Attribution License (https://creativecommons.org/licenses/by/4.0), which permits unrestricted use, distribution and reproduction in any medium provided that the original work is properly attributed.
spellingShingle Research Article
Kujawski, Satu
Crespo, Cátia
Luz, Marta
Yuan, Michaela
Winkler, Sylke
Knust, Elisabeth
Loss of Crb2b-lf leads to anterior segment defects in old zebrafish
title Loss of Crb2b-lf leads to anterior segment defects in old zebrafish
title_full Loss of Crb2b-lf leads to anterior segment defects in old zebrafish
title_fullStr Loss of Crb2b-lf leads to anterior segment defects in old zebrafish
title_full_unstemmed Loss of Crb2b-lf leads to anterior segment defects in old zebrafish
title_short Loss of Crb2b-lf leads to anterior segment defects in old zebrafish
title_sort loss of crb2b-lf leads to anterior segment defects in old zebrafish
topic Research Article
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC7044448/
https://www.ncbi.nlm.nih.gov/pubmed/31988089
http://dx.doi.org/10.1242/bio.047555
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