4.5 Article

Mutagenesis and phenotyping resources in zebrafish for studying development and human disease

Journal

BRIEFINGS IN FUNCTIONAL GENOMICS
Volume 13, Issue 2, Pages 82-94

Publisher

OXFORD UNIV PRESS
DOI: 10.1093/bfgp/elt042

Keywords

zebrafish; mutagenesis; phenotyping; resources; knockouts

Funding

  1. Intramural Research Program of the National Human Genome Research Institute, National Institutes of Health

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The zebrafish (Danio rerio) is an important model organism for studying development and human disease. The zebrafish has an excellent reference genome and the functions of hundreds of genes have been tested using both forward and reverse genetic approaches. Recent years have seen an increasing number of large-scale mutagenesis projects and the number of mutants or gene knockouts in zebrafish has increased rapidly, including for the first time conditional knockout technologies. In addition, targeted mutagenesis techniques such as zinc finger nucleases, transcription activator-like effector nucleases and clustered regularly interspaced short sequences (CRISPR) or CRISPR-associated (Cas), have all been shown to effectively target zebrafish genes as well as the first reported germline homologous recombination, further expanding the utility and power of zebrafish genetics. Given this explosion of mutagenesis resources, it is now possible to perform systematic, high-throughput phenotype analysis of all zebrafish gene knockouts.

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