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

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 pot...

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Autores principales: Vaz, Raquel, Hofmeister, Wolfgang, Lindstrand, Anna
Formato: Online Artículo Texto
Lenguaje:English
Publicado: MDPI 2019
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC6471844/
https://www.ncbi.nlm.nih.gov/pubmed/30875831
http://dx.doi.org/10.3390/ijms20061296
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author Vaz, Raquel
Hofmeister, Wolfgang
Lindstrand, Anna
author_facet Vaz, Raquel
Hofmeister, Wolfgang
Lindstrand, Anna
author_sort Vaz, Raquel
collection PubMed
description 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.
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spelling pubmed-64718442019-04-26 Zebrafish Models of Neurodevelopmental Disorders: Limitations and Benefits of Current Tools and Techniques Vaz, Raquel Hofmeister, Wolfgang Lindstrand, Anna Int J Mol Sci Review 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. MDPI 2019-03-14 /pmc/articles/PMC6471844/ /pubmed/30875831 http://dx.doi.org/10.3390/ijms20061296 Text en © 2019 by the authors. Licensee MDPI, Basel, Switzerland. This article is an open access article distributed under the terms and conditions of the Creative Commons Attribution (CC BY) license (http://creativecommons.org/licenses/by/4.0/).
spellingShingle Review
Vaz, Raquel
Hofmeister, Wolfgang
Lindstrand, Anna
Zebrafish Models of Neurodevelopmental Disorders: Limitations and Benefits of Current Tools and Techniques
title Zebrafish Models of Neurodevelopmental Disorders: Limitations and Benefits of Current Tools and Techniques
title_full Zebrafish Models of Neurodevelopmental Disorders: Limitations and Benefits of Current Tools and Techniques
title_fullStr Zebrafish Models of Neurodevelopmental Disorders: Limitations and Benefits of Current Tools and Techniques
title_full_unstemmed Zebrafish Models of Neurodevelopmental Disorders: Limitations and Benefits of Current Tools and Techniques
title_short Zebrafish Models of Neurodevelopmental Disorders: Limitations and Benefits of Current Tools and Techniques
title_sort zebrafish models of neurodevelopmental disorders: limitations and benefits of current tools and techniques
topic Review
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC6471844/
https://www.ncbi.nlm.nih.gov/pubmed/30875831
http://dx.doi.org/10.3390/ijms20061296
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