Abstract
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.
Highlights
The zebrafish, Danio rerio, is a powerful model in which to study inherited diseases[1,2]
The disease pathogenesis of nephronophthisis and related ciliopathy (NPHP-RC) is intimately related to primary ciliary structure and function
The Kupffer’s vesicle (KV) is a simple yet accessible structure that is ideal for investigating cilia structure and function, and their role in inherited human ciliopathy syndromes
Summary
Using zebrafish to study the function of nephronophthisis and related ciliopathy genes [version 1; peer review: 1 approved, 2 approved with reservations]. Ramsbottom[1], Veronica Sammut[1], Frances E. P. Hughes[1], John A.
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