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Nat Commun 2021 Jan 27;121:612. doi: 10.1038/s41467-021-20902-4.
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The molecular dynamics of subdistal appendages in multi-ciliated cells.

Ryu H , Lee H , Lee J , Noh H , Shin M , Kumar V , Hong S , Kim J , Park S .

The motile cilia of ependymal cells coordinate their beats to facilitate a forceful and directed flow of cerebrospinal fluid (CSF). Each cilium originates from a basal body with a basal foot protruding from one side. A uniform alignment of these basal feet is crucial for the coordination of ciliary beating. The process by which the basal foot originates from subdistal appendages of the basal body, however, is unresolved. Here, we show FGFR1 Oncogene Partner (FOP) is a useful marker for delineating the transformation of a circular, unpolarized subdistal appendage into a polarized structure with a basal foot. Ankyrin repeat and SAM domain-containing protein 1A (ANKS1A) interacts with FOP to assemble region I of the basal foot. Importantly, disruption of ANKS1A reduces the size of region I. This produces an unstable basal foot, which disrupts rotational polarity and the coordinated beating of cilia in young adult mice. ANKS1A deficiency also leads to severe degeneration of the basal foot in aged mice and the detachment of cilia from their basal bodies. This role of ANKS1A in the polarization of the basal foot is evolutionarily conserved in vertebrates. Thus, ANKS1A regulates FOP to build and maintain the polarity of subdistal appendages.

PubMed ID: 33504787
Article link: Nat Commun

Genes referenced: acvr1 ank1 anks1a cdca5 cdk20 cep164 cep43 cntrl fgfr1 fgfr1op2 grap2 odf2 slc7a5 tgfbi Upk3b
GO keywords: ciliary basal body organization [+]
Morpholinos: anks1a MO1

Phenotypes: Xla Wt + anks1a MO (Fig. 8 a) [+]

Article Images: [+] show captions
References [+] :
Boisvieux-Ulrich, The orientation of ciliary basal bodies in quail oviduct is related to the ciliary beating cycle commencement. 1985, Pubmed