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建模复杂的与年龄相关的眼病。

Modeling complex age-related eye disease.

机构信息

John A. Moran Eye Center, University of Utah, Salt Lake City, UT, USA.

John A. Moran Eye Center, University of Utah, Salt Lake City, UT, USA; Biomedical Engineering, University of Utah, Salt Lake City, UT, USA.

出版信息

Prog Retin Eye Res. 2024 May;100:101247. doi: 10.1016/j.preteyeres.2024.101247. Epub 2024 Feb 15.

Abstract

Modeling complex eye diseases like age-related macular degeneration (AMD) and glaucoma poses significant challenges, since these conditions depend highly on age-related changes that occur over several decades, with many contributing factors remaining unknown. Although both diseases exhibit a relatively high heritability of >50%, a large proportion of individuals carrying AMD- or glaucoma-associated genetic risk variants will never develop these diseases. Furthermore, several environmental and lifestyle factors contribute to and modulate the pathogenesis and progression of AMD and glaucoma. Several strategies replicate the impact of genetic risk variants, pathobiological pathways and environmental and lifestyle factors in AMD and glaucoma in mice and other species. In this review we will primarily discuss the most commonly available mouse models, which have and will likely continue to improve our understanding of the pathobiology of age-related eye diseases. Uncertainties persist whether small animal models can truly recapitulate disease progression and vision loss in patients, raising doubts regarding their usefulness when testing novel gene or drug therapies. We will elaborate on concerns that relate to shorter lifespan, body size and allometries, lack of macula and a true lamina cribrosa, as well as absence and sequence disparities of certain genes and differences in their chromosomal location in mice. Since biological, rather than chronological, age likely predisposes an organism for both glaucoma and AMD, more rapidly aging organisms like small rodents may open up possibilities that will make research of these diseases more timely and financially feasible. On the other hand, due to the above-mentioned anatomical and physiological features, as well as pharmacokinetic and -dynamic differences small animal models are not ideal to study the natural progression of vision loss or the efficacy and safety of novel therapies. In this context, we will also discuss the advantages and pitfalls of alternative models that include larger species, such as non-human primates and rabbits, patient-derived retinal organoids, and human organ donor eyes.

摘要

模拟像年龄相关性黄斑变性(AMD)和青光眼这样的复杂眼病具有很大的挑战性,因为这些疾病高度依赖于几十年来发生的与年龄相关的变化,许多相关因素仍然未知。尽管这两种疾病的遗传率都相对较高(>50%),但携带 AMD 或青光眼相关遗传风险变异的大多数个体都不会患上这些疾病。此外,一些环境和生活方式因素也会促进和调节 AMD 和青光眼的发病机制和进展。一些策略在 AMD 和青光眼的小鼠和其他物种中复制了遗传风险变异、病理生物学途径以及环境和生活方式因素的影响。在这篇综述中,我们将主要讨论最常用的小鼠模型,这些模型已经并将继续提高我们对与年龄相关的眼部疾病的病理生物学的理解。目前仍存在一些不确定性,即小型动物模型是否真的可以重现患者的疾病进展和视力丧失,这对它们在测试新型基因或药物疗法时的有用性提出了质疑。我们将详细阐述与小型动物模型相关的一些担忧,包括寿命较短、体型和比例差异、缺乏黄斑和真正的筛板、某些基因的缺失和序列差异以及它们在染色体上位置的差异。由于生物年龄而不是实际年龄可能使机体容易患上青光眼和 AMD,因此像小型啮齿动物这样的衰老速度更快的机体可能会为这些疾病的研究提供更多的机会,使研究更加及时和经济可行。另一方面,由于上述解剖学和生理学特征以及药代动力学和药效学差异,小型动物模型并不适合研究视力丧失的自然进展或新型疗法的疗效和安全性。在这种情况下,我们还将讨论包括较大物种(如非人类灵长类动物和兔子)、患者来源的视网膜类器官以及人供体眼球在内的替代模型的优缺点。

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