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Published online October 22, 2007
doi:10.1083/jcb.200707181
The Journal of Cell Biology, Vol. 179, No. 2, 321-330
The Rockefeller University Press, 0021-9525 $30.00
© 2007 Graser et al.
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Article

 Cep164, a novel centriole appendage protein required for primary cilium formation

Susanne Graser1, York-Dieter Stierhof2, Sébastien B. Lavoie1, Oliver S. Gassner1, Stefan Lamla1, Mikael Le Clech1, and Erich A. Nigg1

1 Department of Cell Biology, Max Planck Institute of Biochemistry, Martinsried D-82152, Germany
2 Electron Microscopy Unit, Center for Plant Molecular Biology, University of Tübingen, Tübingen D-72076, Germany

Correspondence to E. Nigg: nigg{at}biochem.mpg.de

Primary cilia (PC) function as microtubule-based sensory antennae projecting from the surface of many eukaryotic cells. They play important roles in mechano- and chemosensory perception and their dysfunction is implicated in developmental disorders and severe diseases. The basal body that functions in PC assembly is derived from the mature centriole, a component of the centrosome. Through a small interfering RNA screen we found several centrosomal proteins (Ceps) to be involved in PC formation. One newly identified protein, Cep164, was indispensable for PC formation and hence characterized in detail. By immunogold electron microscopy, Cep164 could be localized to the distal appendages of mature centrioles. In contrast to ninein and Cep170, two components of subdistal appendages, Cep164 persisted at centrioles throughout mitosis. Moreover, the localizations of Cep164 and ninein/Cep170 were mutually independent during interphase. These data implicate distal appendages in PC formation and identify Cep164 as an excellent marker for these structures.

Abbreviations used in this paper: ALMS, Alstrom syndrome; BBS, Bardet-Biedl syndrome; Cep, centrosomal protein; C-Nap1, centrosomal Nek2-associated protein 1; IF, immunofluorescence; IFT, intraflagellar transport; immuno-EM, immunogold electron microscopy; Odf, outer dense fiber; PC, primary cilium; qRT-PCR, quantitative real-time PCR.


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