|ZFIN ID: ZDB-PUB-111012-15|
Zebrafish assays of ciliopathies
Zaghloul, N.A., and Katsanis, N.
|Source:||Methods in cell biology 105: 257-272 (Chapter)|
|Registered Authors:||Katsanis, Nicholas, Zaghloul, Norann A.|
|Keywords:||ciliary, dysfunction, functionality, gastrulation, microtubule, transduction|
|PubMed:||21951534 Full text @ Meth. Cell. Biol.|
Zaghloul, N.A., and Katsanis, N. (2011) Zebrafish assays of ciliopathies. Methods in cell biology. 105:257-272.
ABSTRACTIn 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.
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