Mice Lacking Gpr179 with Complete Congenital Stationary Night Blindness Are a Good Model for Myopia

Baptiste Wilmet; Jacques Callebert; Robert Duvoisin; Ruben Goulet; Christophe Tourain; Christelle Michiels; Helen Frederiksen; Frank Schaeffel; Olivier Marre; José Alain Sahel; Isabelle Audo; Serge Picaud; Christina Zeitz

doi:10.3390/ijms24010219

Mice Lacking Gpr179 with Complete Congenital Stationary Night Blindness Are a Good Model for Myopia

Baptiste Wilmet, Jacques Callebert, Robert Duvoisin, Ruben Goulet, Christophe Tourain, Christelle Michiels, Helen Frederiksen, Frank Schaeffel, Olivier Marre, José Alain Sahel, Isabelle Audo, Serge Picaud, Christina Zeitz

Research output: Contribution to journal › Article › peer-review

2 Scopus citations

Abstract

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.

Original language	English (US)
Article number	219
Journal	International journal of molecular sciences
Volume	24
Issue number	1
DOIs	https://doi.org/10.3390/ijms24010219
State	Published - Jan 2023
Externally published	Yes

Keywords

CSNB
GPR179
ON-bipolar cells
dopamine
myopia
refractometry

ASJC Scopus subject areas

Catalysis
Molecular Biology
Spectroscopy
Computer Science Applications
Physical and Theoretical Chemistry
Organic Chemistry
Inorganic Chemistry

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Wilmet, B., Callebert, J., Duvoisin, R., Goulet, R., Tourain, C., Michiels, C., Frederiksen, H., Schaeffel, F., Marre, O., Sahel, J. A., Audo, I., Picaud, S., & Zeitz, C. (2023). Mice Lacking Gpr179 with Complete Congenital Stationary Night Blindness Are a Good Model for Myopia. International journal of molecular sciences, 24(1), Article 219. https://doi.org/10.3390/ijms24010219

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abstract = "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.",

keywords = "CSNB, GPR179, ON-bipolar cells, dopamine, myopia, refractometry",

author = "Baptiste Wilmet and Jacques Callebert and Robert Duvoisin and Ruben Goulet and Christophe Tourain and Christelle Michiels and Helen Frederiksen and Frank Schaeffel and Olivier Marre and Sahel, {Jos{\'e} Alain} and Isabelle Audo and Serge Picaud and Christina Zeitz",

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AU - Wilmet, Baptiste

AU - Callebert, Jacques

AU - Duvoisin, Robert

AU - Goulet, Ruben

AU - Tourain, Christophe

AU - Michiels, Christelle

AU - Frederiksen, Helen

AU - Schaeffel, Frank

AU - Marre, Olivier

AU - Sahel, José Alain

AU - Audo, Isabelle

AU - Picaud, Serge

AU - Zeitz, Christina

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N2 - 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.

AB - 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.

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Mice Lacking Gpr179 with Complete Congenital Stationary Night Blindness Are a Good Model for Myopia

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