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

Mice Lacking Gpr179 with Complete Congenital Stationary Night Blindness Are a Good Model for Myopia.
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缺乏Gpr179的完全先天性静止性夜盲小鼠是近视的良好模型。

DOI:
10.3390/ijms24010219
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发表时间:
2022-12-22
影响因子:
5.6
通讯作者:
Zeitz, Christina
Zeitz, Christina
中科院分区:
生物学2区
文献类型:
--
作者:
Wilmet, Baptiste;Callebert, Jacques;Duvoisin, Robert;Goulet, Ruben;Tourain, Christophe;Michiels, Christelle;Frederiksen, Helen;Schaeffel, Frank;Marre, Olivier;Sahel, Jose Alain;Audo, Isabelle;Picaud, Serge;Zeitz, Christina

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GPR179 突变是常染色体隐性遗传性完全先天性静止性夜盲症 (cCSNB) 的最常见原因之一。这种视网膜疾病的特点是患者视力模糊和夜视受损,并伴有其他眼部症状,包括高度近视。 cCSNB 是由光感受器到 ON-双极细胞的信号传输完全丢失引起的。在这项研究中,我们假设 Gpr179 的缺乏和随后的 ON 通路受损可能导致 cCSNB 小鼠模型出现近视特征。使用超高效液相色谱法,我们发现与 Gpr179+/+ 小鼠相比,成年 Gpr179−/− 小鼠的视网膜多巴胺和 3,4-二羟基苯乙酸显着减少。多巴胺能系统的这种改变被认为与晶状体引起的近视的易感性增加相关,但不影响自然屈光发育。总而言之,我们的数据添加了一种新的近视模型,可用于确定治疗干预措施。
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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