Modelling inborn errors of metabolism in zebrafish

Wager, Kim ORCID: https://orcid.org/0000-0001-8107-9600, Mahmood, Fahad and Russell, Claire 2014. Modelling inborn errors of metabolism in zebrafish. Journal of Inherited Metabolic Disease 37 (4) , pp. 483-495. 10.1007/s10545-014-9696-5

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Abstract

The majority of human inborn errors of metabolism are fatal multisystem disorders that lack proper treatment and have a poorly understood mechanistic basis. Novel technologies are required to address this issue, and the use of zebrafish to model these diseases is an emerging field. Here we present the published zebrafish models of inborn metabolic diseases, discuss their validity, and review the novel mechanistic insights that they have provided. We also review the available methods for creating and studying zebrafish disease models, advantages and disadvantages of using this model organism, and successful examples of the use of zebrafish for drug discovery and development. Using a zebrafish to model inborn errors of metabolism in vivo, although still in its infancy, shows promise for a deeper understanding of disease pathomechanisms, onset, and progression, and also for the development of specific therapies.

Item Type:	Article
Date Type:	Publication
Status:	Published
Schools:	Biosciences
Subjects:	Q Science > QH Natural history > QH426 Genetics R Medicine > RC Internal medicine R Medicine > RJ Pediatrics
Publisher:	Springer
ISSN:	0141-8955
Last Modified:	25 Oct 2022 09:39
URI:	https://orca.cardiff.ac.uk/id/eprint/59495

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