Ktu/PF13 is required for cytoplasmic pre-assembly of axonemal dyneins

Heymut Omran (), Daisuke Kobayashi, Heike Olbrich, Tatsuya Tsukahara, Niki T. Loges, Haruo Hagiwara, Qi Zhang, Gerard Leblond, Eileen O’Toole, Chikako Hara, Hideaki Mizuno, Hiroyuki Kawano, Manfred Fliegauf, Toshiki Yagi, Sumito Koshida, Atsushi Miyawaki, Hanswalter Zentgraf, Horst Seithe, Richard Reinhardt, Yoshinori Watanabe, Ritsu Kamiya, David R. Mitchell () and Hiroyuki Takeda ()
Additional contact information
Heymut Omran: University Hospital Freiburg Mathildenstraße 1
Daisuke Kobayashi: Graduate School of Science, University of Tokyo
Heike Olbrich: University Hospital Freiburg Mathildenstraße 1
Tatsuya Tsukahara: Institute of Molecular and Cellular Biosciences, and Graduate Program in Biophysics and Biochemistry, Graduate School of Science, University of Tokyo
Niki T. Loges: University Hospital Freiburg Mathildenstraße 1
Haruo Hagiwara: Gunma University Graduate School of Medicine, Maebashi, Gunma 371-8511, Japan
Qi Zhang: SUNY Upstate Medical University, Syracuse, New York 13210-1605, USA
Gerard Leblond: SUNY Upstate Medical University, Syracuse, New York 13210-1605, USA
Eileen O’Toole: Cellular and Developmental Biology, University of Colorado, Boulder, Colorado 80309-0347, USA
Chikako Hara: Laboratory for Cell Function Dynamics, Advanced Technology Development Group, Brain Science Institute, RIKEN, Wako, Saitama 351-0198, Japan
Hideaki Mizuno: Laboratory for Cell Function Dynamics, Advanced Technology Development Group, Brain Science Institute, RIKEN, Wako, Saitama 351-0198, Japan
Hiroyuki Kawano: Laboratory for Cell Function Dynamics, Advanced Technology Development Group, Brain Science Institute, RIKEN, Wako, Saitama 351-0198, Japan
Manfred Fliegauf: University Hospital Freiburg Mathildenstraße 1
Toshiki Yagi: Graduate School of Science, University of Tokyo
Sumito Koshida: Graduate School of Science, University of Tokyo
Atsushi Miyawaki: Laboratory for Cell Function Dynamics, Advanced Technology Development Group, Brain Science Institute, RIKEN, Wako, Saitama 351-0198, Japan
Hanswalter Zentgraf: German Cancer Research Center
Horst Seithe: Klinik für Kinder und Jugendliche, Klinikum Nürnberg Süd, Breslauer Straße 201, 90471 Nürnberg, Germany
Richard Reinhardt: Max-Planck-Institut für molekulare Genetik
Yoshinori Watanabe: Institute of Molecular and Cellular Biosciences, and Graduate Program in Biophysics and Biochemistry, Graduate School of Science, University of Tokyo
Ritsu Kamiya: Graduate School of Science, University of Tokyo
David R. Mitchell: SUNY Upstate Medical University, Syracuse, New York 13210-1605, USA
Hiroyuki Takeda: Graduate School of Science, University of Tokyo

Nature, 2008, vol. 456, issue 7222, 611-616

Abstract: Abstract Cilia and flagella are highly conserved organelles that have diverse roles in cell motility and sensing extracellular signals. Motility defects in cilia and flagella often result in primary ciliary dyskinesia. However, the mechanisms underlying cilia formation and function, and in particular the cytoplasmic assembly of dyneins that power ciliary motility, are only poorly understood. Here we report a new gene, kintoun (ktu), involved in this cytoplasmic process. This gene was first identified in a medaka mutant, and found to be mutated in primary ciliary dyskinesia patients from two affected families as well as in the pf13 mutant of Chlamydomonas. In the absence of Ktu/PF13, both outer and inner dynein arms are missing or defective in the axoneme, leading to a loss of motility. Biochemical and immunohistochemical studies show that Ktu/PF13 is one of the long-sought proteins involved in pre-assembly of dynein arm complexes in the cytoplasm before intraflagellar transport loads them for the ciliary compartment.

Date: 2008
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DOI: 10.1038/nature07471

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