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斑马鱼神经发育障碍模型:当前工具和技术的局限性和优势。

Zebrafish Models of Neurodevelopmental Disorders: Limitations and Benefits of Current Tools and Techniques.

机构信息

Department of Molecular Medicine and Surgery, Center for Molecular Medicine, Karolinska Institutet, 171 76 Stockholm, Sweden.

Laboratory of Molecular and Cellular Cardiology, Department of Clinical Biochemistry and Pharmacology, Odense University Hospital, 5000 Odense, Denmark and the Novo Nordisk Foundation for Stem cell Biology (Danstem), University of Copenhagen, 2200 Copenhagen, Denmark.

出版信息

Int J Mol Sci. 2019 Mar 14;20(6):1296. doi: 10.3390/ijms20061296.

Abstract

For the past few years there has been an exponential increase in the use of animal models to confirm the pathogenicity of candidate disease-causing genetic variants found in patients. One such animal model is the zebrafish. Despite being a non-mammalian animal, the zebrafish model has proven its potential in recapitulating the phenotypes of many different human genetic disorders. This review will focus on recent advances in the modeling of neurodevelopmental disorders in zebrafish, covering aspects from early brain development to techniques used for modulating gene expression, as well as how to best characterize the resulting phenotypes. We also review other existing models of neurodevelopmental disorders, and the current efforts in developing and testing compounds with potential therapeutic value.

摘要

在过去的几年中,人们越来越多地使用动物模型来确认在患者中发现的候选致病基因突变的致病性。斑马鱼就是这样一种动物模型。尽管斑马鱼是一种非哺乳动物,但它在重现许多不同人类遗传疾病的表型方面已经证明了其潜力。这篇综述将重点介绍斑马鱼神经发育障碍模型的最新进展,涵盖从早期大脑发育到用于调节基因表达的技术,以及如何最好地描述由此产生的表型等方面。我们还回顾了其他现有的神经发育障碍模型,以及目前在开发和测试具有潜在治疗价值的化合物方面的努力。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/8b81/6471844/ca0656e800cd/ijms-20-01296-g001.jpg

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