PUBLICATION
Ccdc57 is required for straightening the body axis by regulating ciliary motility in the brain ventricle of zebrafish
- Authors
- Li, L., Li, J., Ou, Y., Wu, J., Li, H., Wang, X., Tang, L., Dai, X., Yang, C., Wei, Z., Yin, Z., Shu, Y.
- ID
- ZDB-PUB-230122-6
- Date
- 2023
- Source
- Journal of genetics and genomics = Yi chuan xue bao 50(4): 253-263 (Journal)
- Registered Authors
- Yin, Zhan
- Keywords
- CSF, ccdc57, cilia, epinephrine, scoliosis, urotensin
- MeSH Terms
-
- Animals
- Brain/metabolism
- Cilia/metabolism
- Scoliosis*/metabolism
- Tubulin/metabolism
- Zebrafish*/metabolism
- Zebrafish Proteins/genetics
- Zebrafish Proteins/metabolism
- PubMed
- 36669737 Full text @ J. Genet. Genomics
Citation
Li, L., Li, J., Ou, Y., Wu, J., Li, H., Wang, X., Tang, L., Dai, X., Yang, C., Wei, Z., Yin, Z., Shu, Y. (2023) Ccdc57 is required for straightening the body axis by regulating ciliary motility in the brain ventricle of zebrafish. Journal of genetics and genomics = Yi chuan xue bao. 50(4):253-263.
Abstract
Recently, cilia defects have been proposed to contribute to scoliosis. Here, we demonstrate that Ccdc57 plays an essential role in straightening the body axis of zebrafish by regulating ciliary beating in the brain ventricle (BV). Zygotic ccdc57 (Zccdc57) mutant zebrafish developes scoliosis without significant changes in their bone density and calcification, and the maternal-zygotic ccdc57 (MZccdc57) mutant embryos display curved bodies since the long-pec stage. The expression of ccdc57 is enriched in ciliated tissues and immunofluorescence analysis reveals colocalization of Ccdc57-HA with acetylated α-tubulin, implicating it in having a role in ciliary function. Further examination reveals that it is the coordinated cilia beating of multiple cilia bundles (MCB) in the Mzccdc57 mutant embryos that is affected at 48 hours post fertilization (hpf), when the compromised cerebrospinal fluid (CSF) flow and curved body axis have already occurred. Either ccdc57 mRNA injection or epinephrine treatment reverses the spinal curvature in MZccdc57 mutant larvae from ventrally curly to straight or even dorsally curly and significantly upregulates urotensin signaling. This study reveals the role of ccdc57 in maintaining coordinated cilia beating of MCB in the BV.
Genes / Markers
Expression
Phenotype
Mutations / Transgenics
Human Disease / Model
Sequence Targeting Reagents
Fish
Orthology
Engineered Foreign Genes
Mapping