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Using Confocal Analysis of Xenopus laevis to Investigate Modulators of Wnt and Shh Morphogen Gradients
Published on: December 14, 2015
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Ccdc57 regulates cilia and left-right patterning in Xenopus
Binyi Yang1,2, Emily K Mis2, Xianglin Zhou1
1Department of Pulmonary and Critical Care Medicine, Second Xiangya Hospital, Central South University, Changsha, Hunan 410011, China.
Biology Open
|February 27, 2026
Summary
CCDC57 regulates ciliary function crucial for left-right asymmetry during embryonic development. Mutations in CCDC57 are linked to laterality disorders, impacting cilia structure and function.
Area of Science:
- Developmental Biology
- Cell Biology
- Genetics
Background:
- Left-right (LR) asymmetry is established by ciliary fluid flow in the left-right organizer (LRO).
- Disruptions in LR patterning cause laterality disorders, including situs inversus and congenital heart defects.
Purpose of the Study:
- To identify novel regulators of ciliary function and LR patterning.
- To investigate the role of CCDC57 in embryogenesis and its potential link to human laterality disorders.
Main Methods:
- Depletion of ccdc57 using morpholino oligonucleotides (MOs) in Xenopus embryos.
- Analysis of ciliary structure and function in multiciliated cells.
- Assessment of LR marker gene expression (dand5, pitx2c) and cardiac looping.
- Functional validation of patient-derived CCDC57 variants in a Xenopus model.
Main Results:
- CCDC57 depletion caused abnormal cilia and misexpression of LR markers in Xenopus.
- Cardiac looping defects were observed in ccdc57-depleted embryos.
- Patient-derived CCDC57 variants failed to rescue ciliary defects, unlike wild-type CCDC57.
Conclusions:
- CCDC57 is a critical regulator of ciliary function essential for LR patterning.
- CCDC57 dysfunction is implicated in human laterality disorders, highlighting its role in embryonic development.
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