Please use this identifier to cite or link to this item: https://doi.org/10.1242/dev.108209
Title: Systematic discovery of novel ciliary genes through functional genomics in the zebrafish
Authors: Choksi, S.P
Babu, D 
Lau, D
Yu, X
Roy, S 
Keywords: winged helix transcription factor
article
cell differentiation
ciliary dyskinesia
embryo
eukaryotic flagellum
functional genomics
gene control
gene expression
nonhuman
priority journal
spermatogenesis
transgenic zebrafish
upregulation
Ciliary gene screen
Ciliopathy
Foxj1
Motile cilia
Primary ciliary dyskinesia
Zebrafish
Animals
Cilia
Ciliary Motility Disorders
Embryo, Nonmammalian
Forkhead Transcription Factors
Gene Expression Regulation, Developmental
Gene Knockdown Techniques
Genetic Association Studies
Genomics
Green Fluorescent Proteins
Humans
Morpholinos
Organogenesis
Phenotype
Up-Regulation
Zebrafish
Zebrafish Proteins
Issue Date: 2014
Publisher: Company of Biologists Ltd
Citation: Choksi, S.P, Babu, D, Lau, D, Yu, X, Roy, S (2014). Systematic discovery of novel ciliary genes through functional genomics in the zebrafish. Development (Cambridge) 141 (17) : 3410-3419. ScholarBank@NUS Repository. https://doi.org/10.1242/dev.108209
Rights: Attribution 4.0 International
Abstract: Cilia are microtubule-based hair-like organelles that play many important roles in development and physiology, and are implicated in a rapidly expanding spectrum of human diseases, collectively termed ciliopathies. Primary ciliary dyskinesia (PCD), one of the most prevalent of ciliopathies, arises from abnormalities in the differentiation or motility of the motile cilia. Despite their biomedical importance, a methodical functional screen for ciliary genes has not been carried out in any vertebrate at the organismal level. We sought to systematically discover novel motile cilia genes by identifying the genes induced by Foxj1, a winged-helix transcription factor that has an evolutionarily conserved role as the master regulator of motile cilia biogenesis. Unexpectedly, we find that the majority of the Foxj1-induced genes have not been associated with cilia before. To characterize these novel putative ciliary genes, we subjected 50 randomly selected candidates to a systematic functional phenotypic screen in zebrafish embryos. Remarkably, we find that over 60% are required for ciliary differentiation or function, whereas 30% of the proteins encoded by these genes localize to motile cilia. We also show that these genes regulate the proper differentiation and beating of motile cilia. This collection of Foxj1-induced genes will be invaluable for furthering our understanding of ciliary biology, and in the identification of new mutations underlying ciliary disorders in humans. © 2014. Published by The Company of Biologists Ltd.
Source Title: Development (Cambridge)
URI: https://scholarbank.nus.edu.sg/handle/10635/180169
ISSN: 0950-1991
DOI: 10.1242/dev.108209
Rights: Attribution 4.0 International
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