CAMSAP3 is required for mTORC1-dependent ependymal cell growth and lateral ventricle shaping in mouse brains

Kimura, Toshiya; Saito, Hiroko; Kawsaki, Miwa; Takeichi, Masatoshi

doi:10.1101/2020.07.19.211383

Cited by 3 publications

(2 citation statements)

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“…The subapical microtubule network contributes to both morphogenesis and function of epithelia, including such diverse roles as the regulation of cell morphology and adhesion, planar polarity, and positioning of cilia and their coordinated beating (Vladar et al, 2012;Gomez et al, 2016;Herawati et al, 2016;Tateishi et al, 2017;Kimura et al, 2021;Akhmanova and Kapitein, 2022). However, it is unclear what determines the correct organisation of subapical microtubule networks and two competing models have been proposed.…”

Section: Introductionmentioning

confidence: 99%

Deciphering the roles of cell shape and Fat and Dachsous planar polarity in arranging the Drosophila apical microtubule network through quantitative image analysis

Moreno

Hunton

Strutt

et al. 2023

MBoC

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In epithelial cells, planar polarisation of subapical microtubule networks is thought to be important for both breaking cellular symmetry and maintaining the resulting cellular polarity. Studies in the Drosophila pupal wing and other tissues have suggested two alternative mechanisms for specifying network polarity. On one hand mechanical strain and/or cell shape have been implicated as key determinants, on the other the Fat-Dachsous planar polarity pathway has been suggested to be the primary polarising cue. Using quantitative image analysis in the pupal wing, we reassess these models. We found that cell shape was a strong predictor of microtubule organisation in the developing wing epithelium. Conversely Fat-Dachsous polarity cues do not play any direct role in the organisation of the subapical microtubule network, despite being able to weakly recruit the microtubule minus-end capping protein Patronin to cell boundaries. We conclude that any effect of Fat-Dachsous on microtubule polarity is likely to be indirect, via their known ability to regulate cell shape.

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Section: Introductionmentioning

confidence: 99%

Deciphering the roles of cell shape and Fat and Dachsous planar polarity in arranging the Drosophila apical microtubule network through quantitative image analysis

Moreno

Hunton

Strutt

et al. 2023

MBoC

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“…3c-e), Camsap3deficient mice display only mild or no hydrocephalus phenotypes 66 . Two subsequent publications report that Camsap3deficient mice have normal beating cilia in ependymal cells 67 and normal CP formation in oviduct multicilia 68 . These results also echo our proposed redundant and collective effects of Camsaps.…”

Section: Discussionmentioning

confidence: 98%

Wdr47, Camsaps, and Katanin cooperate to generate ciliary central microtubules

et al. 2021

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The axonemal central pair (CP) are non-centrosomal microtubules critical for planar ciliary beat. How they form, however, is poorly understood. Here, we show that mammalian CP formation requires Wdr47, Camsaps, and microtubule-severing activity of Katanin. Katanin severs peripheral microtubules to produce central microtubule seeds in nascent cilia. Camsaps stabilize minus ends of the seeds to facilitate microtubule outgrowth, whereas Wdr47 concentrates Camsaps into the axonemal central lumen to properly position central microtubules. Wdr47 deficiency in mouse multicilia results in complete loss of CP, rotatory beat, and primary ciliary dyskinesia. Overexpression of Camsaps or their microtubule-binding regions induces central microtubules in Wdr47−/− ependymal cells but at the expense of low efficiency, abnormal numbers, and wrong location. Katanin levels and activity also impact the central microtubule number. We propose that Wdr47, Camsaps, and Katanin function together for the generation of non-centrosomal microtubule arrays in polarized subcellular compartments.

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

Saito

Usami

Fujimori

et al. 2021

Preprint

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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, which are linked by a ‘transition zone’. 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, using superresolution microscopy, we show that CAMSAP3, a protein that can stabilize the minus end of a microtubule, concentrates at multiple sites of the cilium-basal body complex, including the upper region of the transition zone or the axonemal basal plate where the central pair of microtubules (CP) terminates. CAMSAP3 dysfunction resulted in loss of the CP and partial distortion of the basal plate, as well as the failure of multicilia to undergo synchronized beating. These findings indicate that CAMSAP3 plays pivotal roles in the formation or stabilization of the CP, and thereby supports the coordinated motion of multicilia in airway epithelial cells.

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CAMSAP3 is required for mTORC1-dependent ependymal cell growth and lateral ventricle shaping in mouse brains

Cited by 3 publications

References 46 publications

Deciphering the roles of cell shape and Fat and Dachsous planar polarity in arranging the Drosophila apical microtubule network through quantitative image analysis

Deciphering the roles of cell shape and Fat and Dachsous planar polarity in arranging the Drosophila apical microtubule network through quantitative image analysis

Wdr47, Camsaps, and Katanin cooperate to generate ciliary central microtubules

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

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