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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.
Afiliação
  • Wilmet B; Sorbonne Université, INSERM, CNRS, Institut de la Vision, 75012 Paris, France.
  • Callebert J; Service of Biochemistry and Molecular Biology, INSERM U942, Hospital Lariboisière, AP-HP, 75010 Paris, France.
  • Duvoisin R; Department of Chemical Physiology & Biochemistry, Oregon Health & Science University, Portland, OR 97239, USA.
  • Goulet R; Sorbonne Université, INSERM, CNRS, Institut de la Vision, 75012 Paris, France.
  • Tourain C; Sorbonne Université, INSERM, CNRS, Institut de la Vision, 75012 Paris, France.
  • Michiels C; Wavefront-Engineering Microscopy Group, Neurophotonics Laboratory, CNRS UMR8250, Paris Descartes University, 75270 Paris, France.
  • Frederiksen H; Sorbonne Université, INSERM, CNRS, Institut de la Vision, 75012 Paris, France.
  • Schaeffel F; Sorbonne Université, INSERM, CNRS, Institut de la Vision, 75012 Paris, France.
  • Marre O; Institute of Molecular and Clinical Ophthalmology Basel (IOB), 4056 Basel, Switzerland.
  • Sahel JA; Section of Neurobiology of the Eye, Ophthalmic Research Institute, University of Tuebingen, 72076 Tuebingen, Germany.
  • Audo I; Zeiss Vision Lab, Ophthalmic Research Institute, University of Tuebingen, 72076 Tuebingen, Germany.
  • Picaud S; Sorbonne Université, INSERM, CNRS, Institut de la Vision, 75012 Paris, France.
  • Zeitz C; Sorbonne Université, INSERM, CNRS, Institut de la Vision, 75012 Paris, France.
Int J Mol Sci ; 24(1)2022 Dec 22.
Article em En | MEDLINE | ID: mdl-36613663
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.
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Texto completo: 1 Coleções: 01-internacional Base de dados: MEDLINE Assunto principal: Cegueira Noturna / Doenças Genéticas Ligadas ao Cromossomo X / Miopia Limite: Animals Idioma: En Revista: Int J Mol Sci Ano de publicação: 2022 Tipo de documento: Article

Texto completo: 1 Coleções: 01-internacional Base de dados: MEDLINE Assunto principal: Cegueira Noturna / Doenças Genéticas Ligadas ao Cromossomo X / Miopia Limite: Animals Idioma: En Revista: Int J Mol Sci Ano de publicação: 2022 Tipo de documento: Article