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Publication : The left-right determinant Inversin is a component of node monocilia and other 9+0 cilia.

First Author  Watanabe D Year  2003
Journal  Development Volume  130
Issue  9 Pages  1725-34
PubMed ID  12642479 Mgi Jnum  J:263077
Mgi Id  MGI:6188083 Doi  10.1242/dev.00407
Citation  Watanabe D, et al. (2003) The left-right determinant Inversin is a component of node monocilia and other 9+0 cilia. Development 130(9):1725-34
abstractText  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 (InvDeltaC) 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.
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