Tracheal motile cilia in mice require CAMSAP3 for the formation of central microtubule pair and coordinated beating

Hiroko Saito, Fumiko Matsukawa-Usami, Toshihiko Fujimori, Toshiya Kimura, Takahiro Ide, Takaki Yamamoto, Tatsuo Shibata, Kenta Onoue, Satoko Okayama, Shigenobu Yonemura, Kazuyo Misaki, Yurina Soba, Yasutaka Kakui, Masamitsu Sato, Mika Toya, Masatoshi Takeichi

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5 Citations (Scopus)


Motile cilia of multiciliated epithelial cells undergo synchronized beating to produce fluid flow along the luminal surface of various organs. Each motile cilium consists of an axoneme and a basal body (BB), which are linked by a “transition zone” (TZ). The axoneme exhibits a characteristic 9+2 microtubule arrangement important for ciliary motion, but how this microtubule system is generated is not yet fully understood. Here we show that calmodulin-regulated spectrin-associated protein 3 (CAMSAP3), a protein that can stabilize the minus-end of a microtubule, concentrates at multiple sites of the cilium–BB complex, including the upper region of the TZ or the axonemal basal plate (BP) where the central pair of microtubules (CP) initiates. CAMSAP3 dysfunction resulted in loss of the CP and partial distortion of the BP, as well as the failure of multicilia to undergo synchronized beating. These findings suggest that CAMSAP3 plays pivotal roles in the formation or stabilization of the CP by localizing at the basal region of the axoneme and thereby supports the coordinated motion of multicilia in airway epithelial cells.

Original languageEnglish
Article numberar12
JournalMolecular Biology of the Cell
Issue number20
Publication statusPublished - Oct 1 2021
Externally publishedYes

ASJC Scopus subject areas

  • Molecular Biology
  • Cell Biology


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