CFAP53 regulates mammalian cilia-type motility patterns through differential localization and recruitment of axonemal dynein components.
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 ependym...
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Online Access: | https://doi.org/10.1371/journal.pgen.1009232 |
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doaj-9a3f285eee0e45349f02127d86aba31e2021-04-21T14:36:51ZengPublic Library of Science (PLoS)PLoS Genetics1553-73901553-74042020-12-011612e100923210.1371/journal.pgen.1009232CFAP53 regulates mammalian cilia-type motility patterns through differential localization and recruitment of axonemal dynein components.Takahiro IdeWang Kyaw TwanHao LuYayoi IkawaLin-Xenia LimNicole HenningerHiromi NishimuraKatsuyoshi TakaokaVijay NarasimhanXiumin YanHidetaka ShiratoriSudipto RoyHiroshi HamadaMotile 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.https://doi.org/10.1371/journal.pgen.1009232 |
collection |
DOAJ |
language |
English |
format |
Article |
sources |
DOAJ |
author |
Takahiro Ide Wang Kyaw Twan Hao Lu Yayoi Ikawa Lin-Xenia Lim Nicole Henninger Hiromi Nishimura Katsuyoshi Takaoka Vijay Narasimhan Xiumin Yan Hidetaka Shiratori Sudipto Roy Hiroshi Hamada |
spellingShingle |
Takahiro Ide Wang Kyaw Twan Hao Lu Yayoi Ikawa Lin-Xenia Lim Nicole Henninger Hiromi Nishimura Katsuyoshi Takaoka Vijay Narasimhan Xiumin Yan Hidetaka Shiratori Sudipto Roy Hiroshi Hamada CFAP53 regulates mammalian cilia-type motility patterns through differential localization and recruitment of axonemal dynein components. PLoS Genetics |
author_facet |
Takahiro Ide Wang Kyaw Twan Hao Lu Yayoi Ikawa Lin-Xenia Lim Nicole Henninger Hiromi Nishimura Katsuyoshi Takaoka Vijay Narasimhan Xiumin Yan Hidetaka Shiratori Sudipto Roy Hiroshi Hamada |
author_sort |
Takahiro Ide |
title |
CFAP53 regulates mammalian cilia-type motility patterns through differential localization and recruitment of axonemal dynein components. |
title_short |
CFAP53 regulates mammalian cilia-type motility patterns through differential localization and recruitment of axonemal dynein components. |
title_full |
CFAP53 regulates mammalian cilia-type motility patterns through differential localization and recruitment of axonemal dynein components. |
title_fullStr |
CFAP53 regulates mammalian cilia-type motility patterns through differential localization and recruitment of axonemal dynein components. |
title_full_unstemmed |
CFAP53 regulates mammalian cilia-type motility patterns through differential localization and recruitment of axonemal dynein components. |
title_sort |
cfap53 regulates mammalian cilia-type motility patterns through differential localization and recruitment of axonemal dynein components. |
publisher |
Public Library of Science (PLoS) |
series |
PLoS Genetics |
issn |
1553-7390 1553-7404 |
publishDate |
2020-12-01 |
description |
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. |
url |
https://doi.org/10.1371/journal.pgen.1009232 |
work_keys_str_mv |
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