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Disease modeling in genetic kidney diseases: zebrafish

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Abstract

Growing numbers of translational genomics studies are based on the highly efficient and versatile zebrafish (Danio rerio) vertebrate model. The increasing types of zebrafish models have improved our understanding of inherited kidney diseases, since they not only display pathophysiological changes but also give us the opportunity to develop and test novel treatment options in a high-throughput manner. New paradigms in inherited kidney diseases have been developed on the basis of the distinct genome conservation of approximately 70 % between zebrafish and humans in terms of existing gene orthologs. Several options are available to determine the functional role of a specific gene or gene sets. Permanent genome editing can be induced via complete gene knockout by using the CRISPR/Cas-system, among others, or via transient modification by using various morpholino techniques. Cross-species rescues succeeding knockdown techniques are employed to determine the functional significance of a target gene or a specific mutation. This article summarizes the current techniques and discusses their perspectives.

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Acknowledgements

Tg(l-fabp:eGFP-DBP) zebrafish were gifts from J. Xie and B. Anand-Apte, Cleveland Clinic, Cleveland, Ohio, USA.

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Authors and Affiliations

  1. Department of Medicine/Nephrology, Hannover Medical School, Hannover, Germany

    Heiko Schenk, Janina Müller-Deile, Mark Kinast & Mario Schiffer

  2. Mount Desert Island Biological Laboratory, Salisbury Cove, Bar Harbor, Me., USA

    Heiko Schenk, Janina Müller-Deile, Mark Kinast & Mario Schiffer

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  1. Heiko Schenk
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  2. Janina Müller-Deile
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  3. Mark Kinast
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  4. Mario Schiffer
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Correspondence to Mario Schiffer.

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Animal work was conducted according to the guidelines of the American Physiologic Society and was approved by the Mount Desert Island Biological Laboratory (Salisbury Cove, Me., USA) Institutional Animal Care and Use Committee (IACUC protocol no. 804). All efforts were made to minimize the number of animals used and their suffering.

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Schenk, H., Müller-Deile, J., Kinast, M. et al. Disease modeling in genetic kidney diseases: zebrafish. Cell Tissue Res 369, 127–141 (2017). https://doi.org/10.1007/s00441-017-2593-0

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