Zebrafish Assays of Ciliopathies

Norann A. Zaghloul*, Elias Nicholas Katsanis

*Corresponding author for this work

Research output: Chapter in Book/Report/Conference proceedingChapter

18 Scopus citations

Abstract

In light of the growing list of human disorders associated with their dysfunction, primary cilia have recently come to attention as being important regulators of developmental signaling pathways and downstream processes. These organelles, present on nearly every vertebrate cell type, are highly conserved structures allowing for study across a range of species. Zebrafish, in particular, have emerged as useful organisms in which to explore the consequences of ciliary dysfunction and to model human ciliopathies. Here, we present a range of useful techniques that allow for investigation of various aspects of ciliary function. The described assays capitalize on the hallmark gastrulation defects associated with ciliary defects as well as relative ease of visualization of cilia in whole-mount embryos. Further, we describe our recently developed assay for querying functionality of human gene variants in live developing embryos. Finally, a current catalog of known zebrafish ciliary mutant lines is included. The techniques presented here provide a basic toolkit for in vivo investigation of both the biological and genetic mechanisms underlying a growing class of human diseases.

Original languageEnglish (US)
Title of host publicationMethods in Cell Biology
PublisherAcademic Press Inc
Pages257-272
Number of pages16
DOIs
StatePublished - 2011

Publication series

NameMethods in Cell Biology
Volume105
ISSN (Print)0091-679X

Keywords

  • Ciliary
  • Dysfunction
  • Functionality
  • Gastrulation
  • Microtubule
  • Transduction

ASJC Scopus subject areas

  • Cell Biology

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