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Using zebrafish to study the function of nephronophthisis and related ciliopathy genes [version 1; referees: 1 approved, 2 approved with reservations]

Lookup NU author(s): Dr Elisa MolinariORCiD, Dr Simon RamsbottomORCiD, Veronica Sammut, Professor John SayerORCiD



This work is licensed under a Creative Commons Attribution 4.0 International License (CC BY 4.0).


© 2018 Molinari E et al. 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.

Publication metadata

Author(s): Molinari E, Ramsbottom SA, Sammut V, Hughes FEP, Sayer JA

Publication type: Article

Publication status: Published

Journal: F1000Research

Year: 2018

Volume: 7

Online publication date: 25/07/2018

Acceptance date: 02/04/2018

Date deposited: 26/09/2018

ISSN (print): 2046-1402

ISSN (electronic): 1759-796X

Publisher: F1000 Research Ltd


DOI: 10.12688/f1000research.15511.1


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