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

F1000Res. 2018 Jul 25:7:1133. doi: 10.12688/f1000research.15511.2. eCollection 2018.

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.

Keywords: Kupffer’s vesicle; acetylated alpha-tubulin; primary cilia; somite.

Publication types

  • Research Support, Non-U.S. Gov't

MeSH terms

  • Animals
  • Disease Models, Animal
  • Gene Deletion*
  • Polycystic Kidney Diseases* / genetics
  • Polycystic Kidney Diseases* / metabolism
  • Polycystic Kidney Diseases* / pathology
  • Zebrafish Proteins* / genetics
  • Zebrafish Proteins* / metabolism
  • Zebrafish* / genetics
  • Zebrafish* / metabolism

Substances

  • Zebrafish Proteins

Grants and funding

This work was supported by Northern Counties Kidney Research Fund, Kidney Research UK (PDF_003_20151124) and the Medical Research Council (MR/M012212/1).