Cargando…

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...

Descripción completa

Detalles Bibliográficos
Autores principales: 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
Formato: Online Artículo Texto
Lenguaje:English
Publicado: MDPI 2022
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
_version_ 1784865489276633088
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
work_keys_str_mv AT wilmetbaptiste micelackinggpr179withcompletecongenitalstationarynightblindnessareagoodmodelformyopia
AT callebertjacques micelackinggpr179withcompletecongenitalstationarynightblindnessareagoodmodelformyopia
AT duvoisinrobert micelackinggpr179withcompletecongenitalstationarynightblindnessareagoodmodelformyopia
AT gouletruben micelackinggpr179withcompletecongenitalstationarynightblindnessareagoodmodelformyopia
AT tourainchristophe micelackinggpr179withcompletecongenitalstationarynightblindnessareagoodmodelformyopia
AT michielschristelle micelackinggpr179withcompletecongenitalstationarynightblindnessareagoodmodelformyopia
AT frederiksenhelen micelackinggpr179withcompletecongenitalstationarynightblindnessareagoodmodelformyopia
AT schaeffelfrank micelackinggpr179withcompletecongenitalstationarynightblindnessareagoodmodelformyopia
AT marreolivier micelackinggpr179withcompletecongenitalstationarynightblindnessareagoodmodelformyopia
AT saheljosealain micelackinggpr179withcompletecongenitalstationarynightblindnessareagoodmodelformyopia
AT audoisabelle micelackinggpr179withcompletecongenitalstationarynightblindnessareagoodmodelformyopia
AT picaudserge micelackinggpr179withcompletecongenitalstationarynightblindnessareagoodmodelformyopia
AT zeitzchristina micelackinggpr179withcompletecongenitalstationarynightblindnessareagoodmodelformyopia