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          Institute: MPI für molekulare Genetik     Collection: Sequencing Group     Display Documents



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ID: 413803.0, MPI für molekulare Genetik / Sequencing Group
Ktu/PF13 is required for cytoplasmic pre-assembly of axonemal dyneins
Authors:Omran, Heymut; Kobayashi, Daisuke; Olbrich, Heike; Tsukahara, Tatsuya; Loges, Niki T.; Hagiwara, Haruo; Zhang, Qi; Leblond, Gerard; O'Toole, Eileen; Hara, Chikako; Mizuno, Hideaki; Kawano, Hiroyuki; Fliegauf, Manfred; Yagi, Toshiki; Koshida, Sumito; Miyawaki, Atsushi; Zentgraf, Hanswalter; Seithe, Horst; Reinhardt, Richard; Watanabe, Yoshinori; Kamiya, Ritsu; Mitchell, David R.; Takeda, Hiroyuki
Language:English
Date of Publication (YYYY-MM-DD):2008-12-04
Title of Journal:Nature
Journal Abbrev.:Nature
Volume:456
Issue / Number:7222
Start Page:611
End Page:616
Copyright:© 2009 Nature Publishing Group, a division of Macmillan Publishers Limited. All Rights Reserved.
Review Status:not specified
Audience:Experts Only
Abstract / Description: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.
Comment of the Author/Creator:Correspondence to: Heymut Omran1,11David R. Mitchell5Hiroyuki Takeda2 Correspondence and requests for materials should be addressed to H.O. (Email: heymut.omran@uniklinik-freiburg.de) or D.R.M. (Email: MitchelD@upstate.edu) and H.T. (Email: htakeda@biol.s.u-tokyo.ac.jp).
External Publication Status:published
Document Type:Article
Communicated by:Richard Reinhardt
Affiliations:MPI für molekulare Genetik
External Affiliations:1.Department of Pediatrics and Adolescent Medicine, University Hospital Freiburg Mathildenstras zlige 1, D-79106 Freiburg, Germany;
2.Department of Biological Sciences, Graduate School of Science, University of Tokyo, Tokyo 113-0033, Japan;
3.Institute of Molecular and Cellular Biosciences, and Graduate Program in Biophysics and Biochemistry, Graduate School of Science, University of Tokyo, Tokyo 113-0032, Japan;
4.Department of Anatomy and Cell Biology, Gunma University Graduate School of Medicine, Maebashi, Gunma 371-8511, Japan;
5.Department of Cell and Developmental Biology, SUNY Upstate Medical University, Syracuse, New York 13210-1605, USA;
6.Department of Molecular, Cellular and Developmental Biology, University of Colorado, Boulder, Colorado 80309-0347, USA;
7.Laboratory for Cell Function Dynamics, Advanced Technology Development Group, Brain Science Institute, RIKEN, Wako, Saitama 351-0198, Japan;
8.Department of Tumor Virology, German Cancer Research Center, D-69120 Heidelberg, Germany;
9.Klinik für Kinder und Jugendliche, Klinikum Nürnberg Süd, Breslauer Strasse 201, 90471 Nürnberg, Germany.
Identifiers:URL:http://www.nature.com/nature/journal/v456/n7222/pd...
DOI:10.1038/nature07471
ISSN:0028-0836
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