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Updated: Jan 9, 2026

Observation of the Ciliary Movement of Choroid Plexus Epithelial Cells Ex Vivo
Published on: July 13, 2015
JHY enables the transition from switchable to fixed ciliary waveforms in metazoan evolution
Qingxia Chen1, Shuxiang Ma2, Hao Liu3
1Ministry of Education-Shanghai Key Laboratory of Children's Environmental Health, Institute of Early Life Health, Xinhua Hospital, Shanghai Jiao Tong University School of Medicine, 200092, Shanghai, China.
WDR47 coordinates JHY and SPEF1 to stabilize central pair microtubules (CP-MTs) in motile cilia. This discovery reveals a novel mechanism for maintaining ciliary function and waveform, crucial for cellular homeostasis.
Area of Science:
- Cell Biology
- Structural Biology
- Evolutionary Biology
Background:
- Motile cilia are essential for movement and homeostasis.
- Central pair microtubules (CP-MTs) drive ciliary beating.
- The stabilization mechanism for CP-MTs is not fully understood.
Purpose of the Study:
- To elucidate the mechanism stabilizing mammalian central pair microtubules (CP-MTs).
- To identify proteins involved in CP-MT stabilization.
- To understand the evolutionary role of these proteins in ciliary function.
Main Methods:
- Proximity interactome analysis of WDR47.
- Identification and characterization of CP-MT-associated proteins.
- Generation and analysis of Jhy-deficient mice.
- Phylogenetic analyses.
Main Results:
- WDR47, JHY, and SPEF1 form a complex that stabilizes CP-MTs.
- WDR47 recruits JHY and SPEF1 to nascent cilia.
- SPEF1 directly interacts with WDR47 and JHY to anchor them to CP-MTs.
- Jhy deficiency disrupts distal CP-MTs and causes abnormal ciliary beating.
- Conserved functions of WDR47 and SPEF1 across species, with JHY emerging in animals.
Conclusions:
- WDR47, JHY, and SPEF1 are crucial for mammalian CP-MT stability.
- This mechanism is vital for maintaining ciliary motility and waveform.
- The evolution of JHY likely enabled more complex ciliary functions in metazoans.
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