CFAP53 regulates mammalian cilia-type motility patterns through differential localization and recruitment of axonemal dynein components.

Autor: Ide T; Laboratory for Organismal Patterning, RIKEN Center for Biosystems Dynamics Research, Kobe, Hyogo, Japan., Twan WK; Laboratory for Organismal Patterning, RIKEN Center for Biosystems Dynamics Research, Kobe, Hyogo, Japan.; Developmental Genetics Group, Graduate School of Frontier Biosciences, Osaka University, Suita, Osaka, Japan., Lu H; Institute of Molecular and Cell Biology, Proteos, Singapore., Ikawa Y; Laboratory for Organismal Patterning, RIKEN Center for Biosystems Dynamics Research, Kobe, Hyogo, Japan., Lim LX; Institute of Molecular and Cell Biology, Proteos, Singapore., Henninger N; Developmental Genetics Group, Graduate School of Frontier Biosciences, Osaka University, Suita, Osaka, Japan., Nishimura H; Laboratory for Organismal Patterning, RIKEN Center for Biosystems Dynamics Research, Kobe, Hyogo, Japan., Takaoka K; Developmental Genetics Group, Graduate School of Frontier Biosciences, Osaka University, Suita, Osaka, Japan., Narasimhan V; Institute of Molecular and Cell Biology, Proteos, Singapore., Yan X; Institute of Biochemistry and Cell Biology, Shanghai Institutes for Biological Sciences, Chinese Academy of Sciences, Shanghai, China., Shiratori H; Developmental Genetics Group, Graduate School of Frontier Biosciences, Osaka University, Suita, Osaka, Japan., Roy S; Institute of Molecular and Cell Biology, Proteos, Singapore.; Department of Biological Sciences, National University of Singapore, Singapore.; Department of Pediatrics, Yong Loo Ling School of Medicine, National University of Singapore, Singapore., Hamada H; Laboratory for Organismal Patterning, RIKEN Center for Biosystems Dynamics Research, Kobe, Hyogo, Japan.; Developmental Genetics Group, Graduate School of Frontier Biosciences, Osaka University, Suita, Osaka, Japan.
Jazyk: angličtina
Zdroj: PLoS genetics [PLoS Genet] 2020 Dec 21; Vol. 16 (12), pp. e1009232. Date of Electronic Publication: 2020 Dec 21 (Print Publication: 2020).
DOI: 10.1371/journal.pgen.1009232
Abstrakt: Motile cilia can beat with distinct patterns, but how motility variations are regulated remain obscure. Here, we have studied the role of the coiled-coil protein CFAP53 in the motility of different cilia-types in the mouse. While node (9+0) cilia of Cfap53 mutants were immotile, tracheal and ependymal (9+2) cilia retained motility, albeit with an altered beat pattern. In node cilia, CFAP53 mainly localized at the base (centriolar satellites), whereas it was also present along the entire axoneme in tracheal cilia. CFAP53 associated tightly with microtubules and interacted with axonemal dyneins and TTC25, a dynein docking complex component. TTC25 and outer dynein arms (ODAs) were lost from node cilia, but were largely maintained in tracheal cilia of Cfap53-/- mice. Thus, CFAP53 at the base of node cilia facilitates axonemal transport of TTC25 and dyneins, while axonemal CFAP53 in 9+2 cilia stabilizes dynein binding to microtubules. Our study establishes how differential localization and function of CFAP53 contributes to the unique motion patterns of two important mammalian cilia-types.
Competing Interests: The authors have declared that no competing interests exist.
Databáze: MEDLINE
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