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

Author

Listed:
  • Heymut Omran

    (University Hospital Freiburg Mathildenstraße 1)

  • Daisuke Kobayashi

    (Graduate School of Science, University of Tokyo
    Present addresses: Department of Anatomy and Developmental Biology, Graduate School of Medical Science, Kyoto Prefectural University of Medicine, Kyoto 602-8566, Japan (D.K.); Structural Biology, Graduate School of Science, Kyoto University, Kyoto 606-8502, Japan (T.Y.); Department of Neurobiology, Schering Plough Research Institute, Kenilworth, New Jersey 07033, USA (Q.Z.).)

  • 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
    Present addresses: Department of Anatomy and Developmental Biology, Graduate School of Medical Science, Kyoto Prefectural University of Medicine, Kyoto 602-8566, Japan (D.K.); Structural Biology, Graduate School of Science, Kyoto University, Kyoto 606-8502, Japan (T.Y.); Department of Neurobiology, Schering Plough Research Institute, Kenilworth, New Jersey 07033, USA (Q.Z.).)

  • 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
    Present addresses: Department of Anatomy and Developmental Biology, Graduate School of Medical Science, Kyoto Prefectural University of Medicine, Kyoto 602-8566, Japan (D.K.); Structural Biology, Graduate School of Science, Kyoto University, Kyoto 606-8502, Japan (T.Y.); Department of Neurobiology, Schering Plough Research Institute, Kenilworth, New Jersey 07033, USA (Q.Z.).)

  • 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)

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.

Suggested Citation

  • 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 & M, 2008. "Ktu/PF13 is required for cytoplasmic pre-assembly of axonemal dyneins," Nature, Nature, vol. 456(7222), pages 611-616, December.
    • Handle: RePEc:nat:nature:v:456:y:2008:i:7222:d:10.1038_nature07471
    DOI: 10.1038/nature07471
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