Drug Discov Today Technol 2018 Aug 01;28:41-52. doi: 10.1016/j.ddtec.2018.07.001.

CRISPR/Cas9 disease models in zebrafish and Xenopus: The genetic renaissance of fish and frogs.

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The speed by which clinical genomics is currently identifying novel potentially pathogenic variants is outperforming the speed by which these can be functionally (genotype-phenotype) annotated in animal disease models. However, over the past few years the emergence of CRISPR/Cas9 as a straight-forward genome editing technology has revolutionized disease modeling in vertebrate non-mammalian model organisms such as zebrafish, medaka and Xenopus. It is now finally possible, by CRISPR/Cas9, to rapidly establish clinically relevant disease models in these organisms. Interestingly, these can provide both cost-effective genotype-phenotype correlations for gene-(variants) and genomic rearrangements obtained from clinical practice, as well as be exploited to perform translational research to improve prospects of disease afflicted patients. In this review, we show an extensive overview of these new CRISPR/Cas9-mediated disease models and provide future prospects that will allow increasingly accurate modeling of human disease in zebrafish, medaka and Xenopus.

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Species referenced: Xenopus
Genes referenced: ccdc40 dnah9 galnt11 ism1 lhx1 ptf1a rbl1 rho rspo2 six6

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