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用于测试晚期疗法的诱导性非人灵长类视网膜变性模型。

Inducible nonhuman primate models of retinal degeneration for testing end-stage therapies.

机构信息

Sorbonne Université, INSERM, CNRS, Institut de la Vision, F-75012 Paris, France.

CHNO des Quinze-Vingts, INSERM-DGOS CIC 1423, F-75012 Paris, France.

出版信息

Sci Adv. 2023 Aug 2;9(31):eadg8163. doi: 10.1126/sciadv.adg8163.

Abstract

The anatomical differences between the retinas of humans and most animal models pose a challenge for testing novel therapies. Nonhuman primate (NHP) retina is anatomically closest to the human retina. However, there is a lack of relevant NHP models of retinal degeneration (RD) suitable for preclinical studies. To address this unmet need, we generated three distinct inducible cynomolgus macaque models of RD. We developed two genetically targeted strategies using optogenetics and CRISPR-Cas9 to ablate rods and mimic rod-cone dystrophy. In addition, we created an acute model by physical separation of the photoreceptors and retinal pigment epithelium using a polymer patch. Among the three models, the CRISPR-Cas9-based approach was the most advantageous model in view of recapitulating disease-specific features and its ease of implementation. The acute model, however, resulted in the fastest degeneration, making it the most relevant model for testing end-stage vision restoration therapies such as stem cell transplantation.

摘要

人类和大多数动物模型的视网膜解剖结构差异给新型治疗方法的测试带来了挑战。非人类灵长类动物(NHP)的视网膜在解剖结构上与人类的视网膜最为接近。然而,目前缺乏适合临床前研究的相关 NHP 视网膜变性(RD)模型。为了满足这一未满足的需求,我们构建了三种不同的诱导性食蟹猴 RD 模型。我们利用光遗传学和 CRISPR-Cas9 开发了两种基因靶向策略,以消除视杆细胞并模拟杆状细胞和锥状细胞营养不良。此外,我们使用聚合物贴片将光感受器和视网膜色素上皮物理分离,从而建立了一种急性模型。在这三种模型中,鉴于 CRISPR-Cas9 方法能够重现特定疾病的特征及其易于实施的特点,该方法是最有利的模型。然而,急性模型导致了最快的变性,使其成为测试干细胞移植等晚期视力恢复治疗方法最相关的模型。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/5d6a/10396314/468d994dff84/sciadv.adg8163-f1.jpg

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