Title: 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 #
Issue Date: Dec-2008
Publisher: Nature Publishing Group
Series Title: Nature vol:456 issue:7222 pages:611-U54
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.
ISSN: 0028-0836
Publication status: published
KU Leuven publication type: IT
Appears in Collections:Molecular Imaging and Photonics
× corresponding author
# (joint) last author

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