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First published online February 21, 2007
doi: 10.1242/10.1242/jcs.03374

Journal of Cell Science 120, 858-867 (2007)
Published by The Company of Biologists 2007
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Research Article

The lissencephaly protein Lis1 is present in motile mammalian cilia and requires outer arm dynein for targeting to Chlamydomonas flagella

Lotte B. Pedersen1,2,*, Panteleimon Rompolas3, Søren T. Christensen1, Joel L. Rosenbaum2 and Stephen M. King3,*

1 Department of Molecular Biology, University of Copenhagen, Universitetsparken 13, DK-2100 Copenhagen OE, Denmark
2 Department of Molecular, Cellular and Developmental Biology, Yale University, New Haven, CT 06520, USA
3 Department of Molecular, Microbial and Structural Biology, University of Connecticut Health Center, Farmington, CT 06030, USA

* Authors for correspondence (e-mail: LBPedersen{at}aki.ku.dk; king{at}neuron.uchc.edu)

Accepted 8 December 2006

Lissencephaly is a developmental brain disorder characterized by a smooth cerebral surface, thickened cortex and misplaced neurons. Classical lissencephaly is caused by mutations in LIS1, which encodes a WD-repeat protein involved in cytoplasmic dynein regulation, mitosis and nuclear migration. Several proteins required for nuclear migration in Aspergillus bind directly to Lis1, including NudC. Mammalian NudC is highly expressed in ciliated epithelia, and localizes to motile cilia in various tissues. Moreover, a NudC ortholog is upregulated upon deflagellation in Chlamydomonas. We found that mammalian Lis1 localizes to motile cilia in trachea and oviduct, but is absent from non-motile primary cilia. Furthermore, we cloned a gene encoding a Lis1-like protein (CrLis1) from Chlamydomonas. CrLis1 is a ~37 kDa protein that contains seven WD-repeat domains, similar to Lis1 proteins from other organisms. Immunoblotting using an anti-CrLis1 antibody revealed that this protein is present in the flagellum and is depleted from flagella of mutants with defective outer dynein arm assembly, including one strain that lacks only the {alpha} heavy chain/light chain 5 thioredoxin complex. Biochemical experiments confirmed that CrLis1 associates with outer dynein arm components and revealed that CrLis1 binds directly to rat NudC. Our results suggest that Lis1 and NudC are present in cilia and flagella and may regulate outer dynein arm activity.

Key words: Chlamydomonas, Cilia, Dynein, Flagella, Lis1, NudC




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[Abstract] [Full Text] [PDF]


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