Ccdc57 is required for straightening the body axis by regulating ciliary motility in the brain ventricle of zebrafish

Lu Li; Juan Li; Yuan Ou; Jiaxin Wu; Huilin Li; Xin Wang; Liying Tang; Xiangyan Dai; Conghui Yang; Zehong Wei; Zhan Yin; Yuqin Shu

doi:10.1016/j.jgg.2022.12.007

Volume 50 Issue 4

Apr. 2023

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Article Navigation > Journal of Genetics and Genomics > 2023 > 50(4): 253-263

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Ccdc57 is required for straightening the body axis by regulating ciliary motility in the brain ventricle of zebrafish

doi: 10.1016/j.jgg.2022.12.007

Lu Li^a,b,
Juan Li^a,b,
Yuan Ou^a,b,
Jiaxin Wu^c,
Huilin Li^a,b,
Xin Wang^a,b,
Liying Tang^b,
Xiangyan Dai^d,
Conghui Yang^a,b,
Zehong Wei^a,b,
Zhan Yin^e,
Yuqin Shu^a,b

a. State Key Laboratory of Developmental Biology of Freshwater Fish, College of Life Sciences, Hunan Normal University, Changsha, Hunan 410081, China;
b. College of Life Sciences, Hunan Normal University, Changsha, Hunan 410081, China;
c. School of Life Sciences, East China Normal University, Shanghai 200241, China;
d. Key Laboratory of Freshwater Fish Reproduction and Development, Ministry of Education, Southwest University, Chongqing 400715, China;
e. State Key Laboratory of Freshwater Ecology and Biotechnology, Institute of Hydrobiology, Chinese Academy of Sciences, Wuhan, Hubei 430072, China

Funds:

We thank Professor Zhu from Xidian University for conducting the micro-CT scanning and analysis, professors Jin and Li from East China Normal University for giving the arl13b-RFP mRNA as a gift. This work was supported by the National Key Research and Development Program, China (2018YFD0900406), the National Natural Science Foundation, China (31802291), and the Natural Science Foundation of Hunan Province (2021JJ40342).

Received Date: 2022-07-19
Accepted Date: 2022-12-31
Rev Recd Date: 2022-12-22
Publish Date: 2023-04-30

Abstract

Abstract

Recently, cilia defects have been proposed to contribute to scoliosis. Here, we demonstrate that coiled-coil domain-containing 57 (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, when the compromised cerebrospinal fluid 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.
- Scoliosis,
- CSF,
- ccdc57,
- Cilia,
- Epinephrine,
- Urotensin

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References(70)

References

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