Journal
MOLECULAR BIOLOGY OF THE CELL
Volume 12, Issue 3, Pages 589-599Publisher
AMER SOC CELL BIOLOGY
DOI: 10.1091/mbc.12.3.589
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- NIDDK NIH HHS [1RO1 DK-55007-01] Funding Source: Medline
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Mutations in Tg737 cause a wide spectrum of phenotypes, including random left-right axis specification, polycystic kidney disease, liver and pancreatic defects, hydrocephalus, and skeletal patterning abnormalities. To further assess the biological function of Tg737 and its role in the mutant pathology, we identified the cell population expressing Tg737 and determined the subcellular localization of its protein product called Polaris. Tg737 expression is associated with cells possessing either motile or immotile cilia and sperm. Similarly, Polaris concentrated just below the apical membrane in the region of the basal bodies and within the cilia or flagellar axoneme. The data suggest that Polaris functions in a ciliogenic pathway or in cilia maintenance, a role supported by the loss of cilia on the ependymal cell layer in ventricles of Tg7370(orpk) brains and by the lack of node cilia in Tg737(Delta2-3 beta Gal) mutants.
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