Zebrafish are an instrumental system for the generation of photoreceptor degeneration models, which can be utilized to determine underlying causes of photoreceptor dysfunction and death, and for the analysis of potential therapeutic compounds, as well as the characterization of regenerative responses. We review the wealth of information from existing zebrafish models of photoreceptor disease, specifically as they relate to currently accepted taxonomic classes of human rod and cone disease. We also highlight that rich, detailed information can be derived from studying photoreceptor development, structure, and function, including behavioural assessments and in vivo imaging of zebrafish. Zebrafish models are available for a diversity of photoreceptor diseases, including cone dystrophies, which are challenging to recapitulate in nocturnal mammalian systems. Newly discovered models of photoreceptor disease and drusenoid deposit formation may not only provide important insights into pathogenesis of disease, but also potential therapeutic approaches. Zebrafish have already shown their use in providing pre-clinical data prior to testing genetic therapies in clinical trials, such as antisense oligonucleotide therapy for Usher syndrome.

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光感受器功能障碍和退化的斑马鱼模型

斑马鱼是生成光感受器变性模型的工具系统，可用于确定光感受器功能障碍和死亡的根本原因，并可用于分析潜在的治疗化合物以及再生反应的表征。我们回顾了现有斑马鱼光感受器疾病模型的大量信息，特别是与目前公认的人类视杆细胞和视锥细胞疾病的分类学类别相关的信息。我们还强调，通过研究光感受器的发育、结构和功能，包括斑马鱼的行为评估和体内成像，可以获得丰富、详细的信息。斑马鱼模型可用于多种光感受器疾病，包括视锥细胞营养不良，这在夜间哺乳动物系统中很难重现。新发现的光感受器疾病和类玻璃膜疣沉积物形成模型不仅可以为疾病的发病机制提供重要的见解，而且还可以提供潜在的治疗方法。斑马鱼已经展示了它们在临床试验中测试基因疗法之前提供临床前数据的用途，例如针对亚瑟综合症的反义寡核苷酸疗法。