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. |