Mice Lacking <i>Gpr179</i> with Complete Congenital Stationary Night Blindness Are a Good Model for Myopia
Mutations in <i>GPR179</i> 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. cC...
| Main Authors: | , , , , , , , , , , , , |
|---|---|
| Format: | Article |
| Language: | English |
| Published: |
MDPI AG
2022-12-01
|
| Series: | International Journal of Molecular Sciences |
| Subjects: | |
| Online Access: | https://www.mdpi.com/1422-0067/24/1/219 |
| _version_ | 1797440465199431680 |
|---|---|
| author | 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 |
| author_facet | 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 |
| author_sort | Baptiste Wilmet |
| collection | DOAJ |
| description | Mutations in <i>GPR179</i> 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 <i>Gpr179</i> 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 <i>Gpr179</i><sup>−/−</sup> mice have a significant decrease in both retinal dopamine and 3,4-dihydroxyphenylacetic acid, compared to <i>Gpr179</i><sup>+/+</sup> 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. |
| first_indexed | 2024-03-09T12:08:34Z |
| format | Article |
| id | doaj.art-4c142a67c30449f4abe5defc1db7bbd5 |
| institution | Directory Open Access Journal |
| issn | 1661-6596 1422-0067 |
| language | English |
| last_indexed | 2024-03-09T12:08:34Z |
| publishDate | 2022-12-01 |
| publisher | MDPI AG |
| record_format | Article |
| series | International Journal of Molecular Sciences |
| spelling | doaj.art-4c142a67c30449f4abe5defc1db7bbd52023-11-30T22:54:42ZengMDPI AGInternational Journal of Molecular Sciences1661-65961422-00672022-12-0124121910.3390/ijms24010219Mice Lacking <i>Gpr179</i> with Complete Congenital Stationary Night Blindness Are a Good Model for MyopiaBaptiste Wilmet0Jacques Callebert1Robert Duvoisin2Ruben Goulet3Christophe Tourain4Christelle Michiels5Helen Frederiksen6Frank Schaeffel7Olivier Marre8José Alain Sahel9Isabelle Audo10Serge Picaud11Christina Zeitz12Sorbonne Université, INSERM, CNRS, Institut de la Vision, 75012 Paris, FranceService of Biochemistry and Molecular Biology, INSERM U942, Hospital Lariboisière, AP-HP, 75010 Paris, FranceDepartment of Chemical Physiology & Biochemistry, Oregon Health & Science University, Portland, OR 97239, USASorbonne Université, INSERM, CNRS, Institut de la Vision, 75012 Paris, FranceSorbonne Université, INSERM, CNRS, Institut de la Vision, 75012 Paris, FranceSorbonne Université, INSERM, CNRS, Institut de la Vision, 75012 Paris, FranceSorbonne Université, INSERM, CNRS, Institut de la Vision, 75012 Paris, FranceInstitute of Molecular and Clinical Ophthalmology Basel (IOB), 4056 Basel, SwitzerlandSorbonne Université, INSERM, CNRS, Institut de la Vision, 75012 Paris, FranceSorbonne Université, INSERM, CNRS, Institut de la Vision, 75012 Paris, FranceSorbonne Université, INSERM, CNRS, Institut de la Vision, 75012 Paris, FranceSorbonne Université, INSERM, CNRS, Institut de la Vision, 75012 Paris, FranceSorbonne Université, INSERM, CNRS, Institut de la Vision, 75012 Paris, FranceMutations in <i>GPR179</i> 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 <i>Gpr179</i> 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 <i>Gpr179</i><sup>−/−</sup> mice have a significant decrease in both retinal dopamine and 3,4-dihydroxyphenylacetic acid, compared to <i>Gpr179</i><sup>+/+</sup> 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.https://www.mdpi.com/1422-0067/24/1/219CSNBmyopiaON-bipolar cellsGPR179refractometrydopamine |
| spellingShingle | 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 Mice Lacking <i>Gpr179</i> with Complete Congenital Stationary Night Blindness Are a Good Model for Myopia International Journal of Molecular Sciences CSNB myopia ON-bipolar cells GPR179 refractometry dopamine |
| title | Mice Lacking <i>Gpr179</i> with Complete Congenital Stationary Night Blindness Are a Good Model for Myopia |
| title_full | Mice Lacking <i>Gpr179</i> with Complete Congenital Stationary Night Blindness Are a Good Model for Myopia |
| title_fullStr | Mice Lacking <i>Gpr179</i> with Complete Congenital Stationary Night Blindness Are a Good Model for Myopia |
| title_full_unstemmed | Mice Lacking <i>Gpr179</i> with Complete Congenital Stationary Night Blindness Are a Good Model for Myopia |
| title_short | Mice Lacking <i>Gpr179</i> with Complete Congenital Stationary Night Blindness Are a Good Model for Myopia |
| title_sort | mice lacking i gpr179 i with complete congenital stationary night blindness are a good model for myopia |
| topic | CSNB myopia ON-bipolar cells GPR179 refractometry dopamine |
| url | https://www.mdpi.com/1422-0067/24/1/219 |
| work_keys_str_mv | AT baptistewilmet micelackingigpr179iwithcompletecongenitalstationarynightblindnessareagoodmodelformyopia AT jacquescallebert micelackingigpr179iwithcompletecongenitalstationarynightblindnessareagoodmodelformyopia AT robertduvoisin micelackingigpr179iwithcompletecongenitalstationarynightblindnessareagoodmodelformyopia AT rubengoulet micelackingigpr179iwithcompletecongenitalstationarynightblindnessareagoodmodelformyopia AT christophetourain micelackingigpr179iwithcompletecongenitalstationarynightblindnessareagoodmodelformyopia AT christellemichiels micelackingigpr179iwithcompletecongenitalstationarynightblindnessareagoodmodelformyopia AT helenfrederiksen micelackingigpr179iwithcompletecongenitalstationarynightblindnessareagoodmodelformyopia AT frankschaeffel micelackingigpr179iwithcompletecongenitalstationarynightblindnessareagoodmodelformyopia AT oliviermarre micelackingigpr179iwithcompletecongenitalstationarynightblindnessareagoodmodelformyopia AT josealainsahel micelackingigpr179iwithcompletecongenitalstationarynightblindnessareagoodmodelformyopia AT isabelleaudo micelackingigpr179iwithcompletecongenitalstationarynightblindnessareagoodmodelformyopia AT sergepicaud micelackingigpr179iwithcompletecongenitalstationarynightblindnessareagoodmodelformyopia AT christinazeitz micelackingigpr179iwithcompletecongenitalstationarynightblindnessareagoodmodelformyopia |