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doi: 10.1242/10.1242/dev.00407
1 Developmental Genetics Group, Graduate School of Frontier Biosciences, Osaka
University, 1-3 Yamada-oka, Suita, Osaka 565-0871, Japan
2 Department of Anatomy and Developmental Biology, Tokyo Women's Medical
University, School of Medicine, 8-1 Kawada-cho, Shinjuku-ku, Tokyo 162-8600,
Japan
* CREST, Japan Science and Technology Corporation (JST)
Present address: Department of Anatomy and Developmental Biology, Kyoto
Prefecture University of Medicine, Kawaramachi-Hirokoji, Kamikyo-ku, Kyoto
602-0841, Japan
Author for correspondence (e-mail:
hamada{at}fbs.osaka-u.ac.jp)
Accepted 13 January 2003
Inversin (Inv), a protein that contains ankyrin repeats, plays a key role
in left-right determination during mammalian embryonic development, but its
precise function remains unknown. Transgenic mice expressing an Inv and green
fluorescent protein (GFP) fusion construct (Inv::GFP) were established to
facilitate characterization of the subcellular localization of Inv. The
Inv::GFP transgene rescued the laterality defects and polycystic
kidney disease of Inv/Inv mice, indicating that the fusion protein is
functional. In transgenic embryos, Inv::GFP protein was detected in the node
monocilia. The fusion protein was also present in other 9+0 monocilia,
including those of kidney epithelial cells and the pituitary gland, but it was
not localized to 9+2 cilia. The N-terminal region of Inv (Inv
C)
including the ankyrin repeats also localized to the node cilia and rescued the
left-right defects of Inv/Inv mutants. Although no obvious
abnormalities were detected in the node monocilia of Inv/Inv embryos,
the laterality defects of such embryos were corrected by an artificial
leftward flow of fluid in the node, suggesting that nodal flow is impaired by
the Inv mutation. These results suggest that the Inv protein
contributes to left-right determination as a component of monocilia in the
node and is essential for the generation of normal nodal flow.
Key words: Cilia, Inv, Left-right asymmetry, Node, Mouse
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