CEP162 is an axoneme-recognition protein promoting ciliary transition zone assembly at the cilia base

Won Jing Wang, Hwee Goon Tay, Rajesh Soni, Geoffrey S. Perumal, Mary G. Goll, Frank P. Macaluso, John M. Asara, Jeffrey D. Amack, Meng Fu Bryan Tsou

Research output: Contribution to journalArticle

41 Citations (Scopus)

Abstract

The transition zone is a specialized compartment found at the base of cilia, adjacent to the centriole distal end, where axonemal microtubules are heavily crosslinked to the surrounding membrane to form a barrier that gates the ciliary compartment. A number of ciliopathy molecules have been found to associate with the transition zone, but factors that directly recognize axonemal microtubules to specify transition zone assembly at the cilia base remain unclear. Here, through quantitative centrosome proteomics, we identify an axoneme-associated protein, CEP162 (KIAA1009), tethered specifically at centriole distal ends to promote transition zone assembly. CEP162 interacts with core transition zone components, and mediates their association with microtubules. Loss of CEP162 arrests ciliogenesis at the stage of transition zone assembly. Abolishing its centriolar tethering, however, allows CEP162 to stay on the growing end of the axoneme and ectopically assemble transition zone components at cilia tips. This generates extra-long cilia with strikingly swollen tips that actively release ciliary contents into the extracellular environment. CEP162 is thus an axoneme-recognition protein pre-tethered at centriole distal ends before ciliogenesis to promote and restrict transition zone formation specifically at the cilia base.

Original languageEnglish (US)
Pages (from-to)591-601
Number of pages11
JournalNature Cell Biology
Volume15
Issue number6
DOIs
StatePublished - Jun 2013

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Axoneme
Cilia
Centrioles
Microtubules
Proteins
Centrosome
Proteomics
Membranes

ASJC Scopus subject areas

  • Cell Biology

Cite this

Wang, W. J., Tay, H. G., Soni, R., Perumal, G. S., Goll, M. G., Macaluso, F. P., ... Bryan Tsou, M. F. (2013). CEP162 is an axoneme-recognition protein promoting ciliary transition zone assembly at the cilia base. Nature Cell Biology, 15(6), 591-601. https://doi.org/10.1038/ncb2739

CEP162 is an axoneme-recognition protein promoting ciliary transition zone assembly at the cilia base. / Wang, Won Jing; Tay, Hwee Goon; Soni, Rajesh; Perumal, Geoffrey S.; Goll, Mary G.; Macaluso, Frank P.; Asara, John M.; Amack, Jeffrey D.; Bryan Tsou, Meng Fu.

In: Nature Cell Biology, Vol. 15, No. 6, 06.2013, p. 591-601.

Research output: Contribution to journalArticle

Wang, WJ, Tay, HG, Soni, R, Perumal, GS, Goll, MG, Macaluso, FP, Asara, JM, Amack, JD & Bryan Tsou, MF 2013, 'CEP162 is an axoneme-recognition protein promoting ciliary transition zone assembly at the cilia base', Nature Cell Biology, vol. 15, no. 6, pp. 591-601. https://doi.org/10.1038/ncb2739
Wang, Won Jing ; Tay, Hwee Goon ; Soni, Rajesh ; Perumal, Geoffrey S. ; Goll, Mary G. ; Macaluso, Frank P. ; Asara, John M. ; Amack, Jeffrey D. ; Bryan Tsou, Meng Fu. / CEP162 is an axoneme-recognition protein promoting ciliary transition zone assembly at the cilia base. In: Nature Cell Biology. 2013 ; Vol. 15, No. 6. pp. 591-601.
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