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Mice Lacking Gpr179 with Complete Congenital Stationary Night Blindness Are a Good Model for Myopia
Mutations in GPR179 are one of the most common causes of autosomal recessive complete congenital stationary night blindness (cCSNB). This retinal disease is characterized in patients by impaired dim and night vision, associated with other ocular symptoms, including high myopia. cCSNB is caused by a...
Autores principales: | , , , , , , , , , , , , |
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Formato: | Online Artículo Texto |
Lenguaje: | English |
Publicado: |
MDPI
2022
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Materias: | |
Acceso en línea: | https://www.ncbi.nlm.nih.gov/pmc/articles/PMC9820543/ https://www.ncbi.nlm.nih.gov/pubmed/36613663 http://dx.doi.org/10.3390/ijms24010219 |
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author | Wilmet, Baptiste Callebert, Jacques Duvoisin, Robert Goulet, Ruben Tourain, Christophe Michiels, Christelle Frederiksen, Helen Schaeffel, Frank Marre, Olivier Sahel, José Alain Audo, Isabelle Picaud, Serge Zeitz, Christina |
author_facet | Wilmet, Baptiste Callebert, Jacques Duvoisin, Robert Goulet, Ruben Tourain, Christophe Michiels, Christelle Frederiksen, Helen Schaeffel, Frank Marre, Olivier Sahel, José Alain Audo, Isabelle Picaud, Serge Zeitz, Christina |
author_sort | Wilmet, Baptiste |
collection | PubMed |
description | Mutations in GPR179 are one of the most common causes of autosomal recessive complete congenital stationary night blindness (cCSNB). This retinal disease is characterized in patients by impaired dim and night vision, associated with other ocular symptoms, including high myopia. cCSNB is caused by a complete loss of signal transmission from photoreceptors to ON-bipolar cells. In this study, we hypothesized that the lack of Gpr179 and the subsequent impaired ON-pathway could lead to myopic features in a mouse model of cCSNB. Using ultra performance liquid chromatography, we show that adult Gpr179(−/−) mice have a significant decrease in both retinal dopamine and 3,4-dihydroxyphenylacetic acid, compared to Gpr179(+/+) mice. This alteration of the dopaminergic system is thought to be correlated with an increased susceptibility to lens-induced myopia but does not affect the natural refractive development. Altogether, our data added a novel myopia model, which could be used to identify therapeutic interventions. |
format | Online Article Text |
id | pubmed-9820543 |
institution | National Center for Biotechnology Information |
language | English |
publishDate | 2022 |
publisher | MDPI |
record_format | MEDLINE/PubMed |
spelling | pubmed-98205432023-01-07 Mice Lacking Gpr179 with Complete Congenital Stationary Night Blindness Are a Good Model for Myopia Wilmet, Baptiste Callebert, Jacques Duvoisin, Robert Goulet, Ruben Tourain, Christophe Michiels, Christelle Frederiksen, Helen Schaeffel, Frank Marre, Olivier Sahel, José Alain Audo, Isabelle Picaud, Serge Zeitz, Christina Int J Mol Sci Article Mutations in GPR179 are one of the most common causes of autosomal recessive complete congenital stationary night blindness (cCSNB). This retinal disease is characterized in patients by impaired dim and night vision, associated with other ocular symptoms, including high myopia. cCSNB is caused by a complete loss of signal transmission from photoreceptors to ON-bipolar cells. In this study, we hypothesized that the lack of Gpr179 and the subsequent impaired ON-pathway could lead to myopic features in a mouse model of cCSNB. Using ultra performance liquid chromatography, we show that adult Gpr179(−/−) mice have a significant decrease in both retinal dopamine and 3,4-dihydroxyphenylacetic acid, compared to Gpr179(+/+) mice. This alteration of the dopaminergic system is thought to be correlated with an increased susceptibility to lens-induced myopia but does not affect the natural refractive development. Altogether, our data added a novel myopia model, which could be used to identify therapeutic interventions. MDPI 2022-12-22 /pmc/articles/PMC9820543/ /pubmed/36613663 http://dx.doi.org/10.3390/ijms24010219 Text en © 2022 by the authors. https://creativecommons.org/licenses/by/4.0/Licensee MDPI, Basel, Switzerland. This article is an open access article distributed under the terms and conditions of the Creative Commons Attribution (CC BY) license (https://creativecommons.org/licenses/by/4.0/). |
spellingShingle | Article Wilmet, Baptiste Callebert, Jacques Duvoisin, Robert Goulet, Ruben Tourain, Christophe Michiels, Christelle Frederiksen, Helen Schaeffel, Frank Marre, Olivier Sahel, José Alain Audo, Isabelle Picaud, Serge Zeitz, Christina Mice Lacking Gpr179 with Complete Congenital Stationary Night Blindness Are a Good Model for Myopia |
title | Mice Lacking Gpr179 with Complete Congenital Stationary Night Blindness Are a Good Model for Myopia |
title_full | Mice Lacking Gpr179 with Complete Congenital Stationary Night Blindness Are a Good Model for Myopia |
title_fullStr | Mice Lacking Gpr179 with Complete Congenital Stationary Night Blindness Are a Good Model for Myopia |
title_full_unstemmed | Mice Lacking Gpr179 with Complete Congenital Stationary Night Blindness Are a Good Model for Myopia |
title_short | Mice Lacking Gpr179 with Complete Congenital Stationary Night Blindness Are a Good Model for Myopia |
title_sort | mice lacking gpr179 with complete congenital stationary night blindness are a good model for myopia |
topic | Article |
url | https://www.ncbi.nlm.nih.gov/pmc/articles/PMC9820543/ https://www.ncbi.nlm.nih.gov/pubmed/36613663 http://dx.doi.org/10.3390/ijms24010219 |
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