Transport of the outer dynein arm complex to cilia requires a cytoplasmic protein Lrrc6

Yasuko Inaba, Kyosuke Shinohara, Yanick Botilde, Ryo Nabeshima, Katsuyoshi Takaoka, Rieko Ajima, Lynda Lamri, Hiroyuki Takeda, Yumiko Saga, Tetsuya Nakamura, Hiroshi Hamada

Research output: Contribution to journalArticlepeer-review

18 Scopus citations


Lrrc6 encodes a cytoplasmic protein that is expressed specifically in cells with motile cilia including the node, trachea and testes of the mice. A mutation of Lrrc6 has been identified in human patients with primary ciliary dyskinesia (PCD). Mutant mice lacking Lrrc6 show typical PCD defects such as hydrocephalus and laterality defects. We found that in the absence of Lrrc6, the morphology of motile cilia remained normal, but their motility was completely lost. The 9 + 2 arrangement of microtubules remained normal in Lrrc6-/- mice, but the outer dynein arms (ODAs), the structures essential for the ciliary beating, were absent from the cilia. In the absence of Lrrc6, ODA proteins such as DNAH5, DNAH9 and IC2, which are assembled in the cytoplasm and transported to the ciliary axoneme, remained in the cytoplasm and were not transported to the ciliary axoneme. The IC2–IC1 interaction, which is the first step of ODA assembly, was normal in Lrrc6−/− mice testes. Our results suggest that ODA proteins may be transported from the cytoplasm to the cilia by an Lrrc6-dependent mechanism.

Original languageEnglish (US)
Pages (from-to)728-739
Number of pages12
JournalGenes to Cells
Issue number7
StatePublished - Jul 1 2016
Externally publishedYes

All Science Journal Classification (ASJC) codes

  • Genetics
  • Cell Biology

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