PUBLICATION
Foxj1 transcription factors are master regulators of the motile ciliogenic program
- Authors
- Yu, X., Ng, C.P., Habacher, H., and Roy, S.
- ID
- ZDB-PUB-081121-12
- Date
- 2008
- Source
- Nature Genetics 40(12): 1445-1453 (Journal)
- Registered Authors
- Roy, Sudipto
- Keywords
- none
- MeSH Terms
-
- Forkhead Transcription Factors/genetics*
- Forkhead Transcription Factors/metabolism
- Cilia/metabolism*
- Zebrafish/embryology*
- Zebrafish/genetics*
- Animals
- Molecular Sequence Data
- Hedgehog Proteins/metabolism
- Signal Transduction
- PubMed
- 19011630 Full text @ Nat. Genet.
Citation
Yu, X., Ng, C.P., Habacher, H., and Roy, S. (2008) Foxj1 transcription factors are master regulators of the motile ciliogenic program. Nature Genetics. 40(12):1445-1453.
Abstract
Motile cilia induce fluid movement through their rhythmic beating activity. In mammals, the transcription factor Foxj1 has been implicated in motile cilia formation. Here we show that a zebrafish Foxj1 homolog, foxj1a, is a target of Hedgehog signaling in the floor plate. Loss of Foxj1a compromises the assembly of motile cilia that decorate floor plate cells. Besides the floor plate, foxj1a is expressed in Kupffer's vesicle and pronephric ducts, where it also promotes ciliary differentiation. We show that Foxj1a activates a constellation of genes essential for motile cilia formation and function, and that its activity is sufficient for ectopic development of cilia that resemble motile cilia. We also document that a paralogous gene, foxj1b, is expressed in the otic vesicle and seems to regulate motile cilia formation in this tissue. Our findings identify a dedicated master regulatory role for Foxj1 in the transcriptional program that controls the production of motile cilia.
Genes / Markers
Expression
Phenotype
Mutations / Transgenics
Human Disease / Model
Sequence Targeting Reagents
Fish
Orthology
Engineered Foreign Genes
Mapping