Using zebrafish to study the function of nephronophthisis and related ciliopathy genes

Zebrafish are a valuable vertebrate model in which to study development and characterize genes involved in cystic kidney disease. Zebrafish embryos and larvae are transparent, allowing non-invasive imaging during their rapid development, which takes place over the first 72 hours post fertilisation....

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Autores principales: Molinari, Elisa, Ramsbottom, Simon A., Sammut, Veronica, Hughes, Frances E. P., Sayer, John A.
Formato: Online Artículo Texto
Lenguaje:English
Publicado: F1000 Research Limited 2018
Materias:
Method Article
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC6127739/
https://www.ncbi.nlm.nih.gov/pubmed/30254740
http://dx.doi.org/10.12688/f1000research.15511.2
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author Molinari, Elisa
Ramsbottom, Simon A.
Sammut, Veronica
Hughes, Frances E. P.
Sayer, John A.
author_facet Molinari, Elisa
Ramsbottom, Simon A.
Sammut, Veronica
Hughes, Frances E. P.
Sayer, John A.
author_sort Molinari, Elisa
collection PubMed
description Zebrafish are a valuable vertebrate model in which to study development and characterize genes involved in cystic kidney disease. Zebrafish embryos and larvae are transparent, allowing non-invasive imaging during their rapid development, which takes place over the first 72 hours post fertilisation. Gene-specific knockdown of nephronophthisis-associated genes leads to ciliary phenotypes which can be assessed in various developmental structures. Here we describe in detail the methods used for imaging cilia within Kupffer’s vesicle to assess nephronophthisis and related ciliopathy phenotypes.
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spelling pubmed-61277392018-09-24 Using zebrafish to study the function of nephronophthisis and related ciliopathy genes Molinari, Elisa Ramsbottom, Simon A. Sammut, Veronica Hughes, Frances E. P. Sayer, John A. F1000Res Method Article Zebrafish are a valuable vertebrate model in which to study development and characterize genes involved in cystic kidney disease. Zebrafish embryos and larvae are transparent, allowing non-invasive imaging during their rapid development, which takes place over the first 72 hours post fertilisation. Gene-specific knockdown of nephronophthisis-associated genes leads to ciliary phenotypes which can be assessed in various developmental structures. Here we describe in detail the methods used for imaging cilia within Kupffer’s vesicle to assess nephronophthisis and related ciliopathy phenotypes. F1000 Research Limited 2018-12-03 /pmc/articles/PMC6127739/ /pubmed/30254740 http://dx.doi.org/10.12688/f1000research.15511.2 Text en Copyright: © 2018 Molinari E et al. http://creativecommons.org/licenses/by/4.0/ This is an open access article distributed under the terms of the Creative Commons Attribution Licence, which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited.
spellingShingle Method Article
Molinari, Elisa
Ramsbottom, Simon A.
Sammut, Veronica
Hughes, Frances E. P.
Sayer, John A.
Using zebrafish to study the function of nephronophthisis and related ciliopathy genes
title Using zebrafish to study the function of nephronophthisis and related ciliopathy genes
title_full Using zebrafish to study the function of nephronophthisis and related ciliopathy genes
title_fullStr Using zebrafish to study the function of nephronophthisis and related ciliopathy genes
title_full_unstemmed Using zebrafish to study the function of nephronophthisis and related ciliopathy genes
title_short Using zebrafish to study the function of nephronophthisis and related ciliopathy genes
title_sort using zebrafish to study the function of nephronophthisis and related ciliopathy genes
topic Method Article
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC6127739/
https://www.ncbi.nlm.nih.gov/pubmed/30254740
http://dx.doi.org/10.12688/f1000research.15511.2
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