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

Omran, H., Kobayashi, D., Olbrich, H., Tsukahara, T., Loges, N. T., Hagiwara, H., et al. (2008). Ktu/PF13 is required for cytoplasmic pre-assembly of axonemal dyneins. Nature, 456(7222), 611-616. doi:10.1038/nature07471.

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Item Permalink: http://hdl.handle.net/11858/00-001M-0000-0010-7E92-3 Version Permalink: http://hdl.handle.net/11858/00-001M-0000-0010-7E93-1
Genre: Journal Article
Alternative Title : Nature

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 Creators:
Omran, Heymut, Author
Kobayashi, Daisuke, Author
Olbrich, Heike, Author
Tsukahara, Tatsuya, Author
Loges, Niki T., Author
Hagiwara, Haruo, Author
Zhang, Qi, Author
Leblond, Gerard, Author
O'Toole, Eileen, Author
Hara, Chikako, Author
Mizuno, Hideaki, Author
Kawano, Hiroyuki, Author
Fliegauf, Manfred, Author
Yagi, Toshiki, Author
Koshida, Sumito, Author
Miyawaki, Atsushi, Author
Zentgraf, Hanswalter, Author
Seithe, Horst, Author
Reinhardt, Richard1, Author              
Watanabe, Yoshinori, Author
Kamiya, Ritsu, AuthorMitchell, David R., AuthorTakeda, Hiroyuki, Author more..
Affiliations:
1High Throughput Technologies, Max Planck Institute for Molecular Genetics, Max Planck Society, ou_1433552              

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 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.

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Language(s): eng - English
 Dates: 2008-12-04
 Publication Status: Published in print
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Title: Nature
  Alternative Title : Nature
Source Genre: Journal
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Pages: - Volume / Issue: 456 (7222) Sequence Number: - Start / End Page: 611 - 616 Identifier: ISSN: 0028-0836