PUBLICATION
FoxF1 is Required for Ciliogenesis and Distribution of Sonic Hedgehog Signaling Components in Cilium
- Authors
- Huang, L., Tjakra, M., Luo, D., Wen, L., Lei, D., Wang, J., Yin, T., Zhang, X., Huang, J., Wang, Y., Wang, G.
- ID
- ZDB-PUB-190406-17
- Date
- 2019
- Source
- Current Molecular Medicine 19(5): 326-334 (Journal)
- Registered Authors
- Huang, Lu, Lei, Daoxi, Luo, Desha, Wang, Guixue, Wang, Yeqi, Wen, Lin, Zhang, Xiaojuan
- Keywords
- FoxF1, Sonic Hedgehog, ciliogenesis, cilium, pronephric cilium, zebrafish mutants
- MeSH Terms
-
- Animals
- Cell Line
- Cilia/genetics*
- Cilia/metabolism*
- Forkhead Transcription Factors/genetics*
- Forkhead Transcription Factors/metabolism*
- Hedgehog Proteins/metabolism*
- Humans
- Mice
- NIH 3T3 Cells
- Organogenesis/genetics*
- Signal Transduction*
- Zebrafish/genetics
- Zebrafish/metabolism
- PubMed
- 30950350 Full text @ Curr. Mol. Med.
Citation
Huang, L., Tjakra, M., Luo, D., Wen, L., Lei, D., Wang, J., Yin, T., Zhang, X., Huang, J., Wang, Y., Wang, G. (2019) FoxF1 is Required for Ciliogenesis and Distribution of Sonic Hedgehog Signaling Components in Cilium. Current Molecular Medicine. 19(5):326-334.
Abstract
Background In vertebrates, cilium is crucial for Hedgehog signaling transduction. Forkhead box transcriptional factor FoxF1 is reported to associate with Sonic Hedgehog (Shh) signaling in many cases. However, the role of FoxF1 in cilium remains unknown. Here, we showed an essential role of FoxF1 in regulation of ciliogenesis and distribution of Shh signaling components in cilium.
Methods NIH/3T3 cells were serum starved for 24h to induce cilium. Meanwhile, shRNA was used to knockdown FoxF1 expression in cells and CRISPR/Cas9 was used to generate FoxF1 zebrafish mutant. The mRNA and protein expression of indicated genes were detected by qRT-PCR and western blot, respectively. Immunofluorescence staining was performed to detect the cilium and Shh components distribution.
Results FoxF1 knockdown decreased the cilium length in NIH/3T3 cells. Meanwhile, disruption of FoxF1 function inhibited the expression of cilium-related genes and caused an abnormal distribution of Shh components in the cilium. Furthermore, homozygous FoxF1 mutants exhibited defective development of pronephric cilium in early zebrafish embryos.
Conclusion Together, our data illustrated that FoxF1 is required for ciliogenesis in vitro and in vivo and for the proper localization of Shh signaling components in cilium.
Genes / Markers
Expression
Phenotype
Mutations / Transgenics
Human Disease / Model
Sequence Targeting Reagents
Fish
Orthology
Engineered Foreign Genes
Mapping