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Publication : Directional ciliary beats across epithelia require Ccdc57-mediated coupling between axonemal orientation and basal body polarity.

First Author  Pan X Year  2024
Journal  Nat Commun Volume  15
Issue  1 Pages  10249
PubMed ID  39592607 Mgi Jnum  J:358923
Mgi Id  MGI:7784568 Doi  10.1038/s41467-024-54766-1
Citation  Pan X, et al. (2024) Directional ciliary beats across epithelia require Ccdc57-mediated coupling between axonemal orientation and basal body polarity. Nat Commun 15(1):10249
abstractText  Motile cilia unify their axonemal orientations (AOs), or beat directions, across epithelia to drive liquid flows. This planar polarity results from cytoskeleton-driven swiveling of basal foot (BF), a basal body (BB) appendage coincident with the AO, in response to regulatory cues. How and when the BF-AO relationship is established, however, are unaddressed. Here, we show that the BF-AO coupling occurs during rotational polarizations of BBs and requires Ccdc57. Ccdc57 localizes on BBs as a rotationally-asymmetric punctum, which polarizes away from the BF in BBs having achieved the rotational polarity to probably fix the BF-AO relationship. Consistently, Ccdc57-deficient ependymal multicilia lack the BF-AO coupling and display directional beats at only single cell level. Ccdc57 (-/-) tracheal multicilia also fail to fully align their BFs. Furthermore, Ccdc57 (-/-) mice manifest severe hydrocephalus, due to impaired cerebrospinal fluid flow, and high mortality. These findings unravel mechanisms governing the planar polarity of epithelial motile cilia.
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