Molecular mechanisms of mechanical signal sensing by motile cilia in vertebrate organogenesis

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Polycystic kidney disease (PKD) is a serious congenital disease in vertebrates, in which epithelial cells in renal tubules abnormally proliferate and become flattened in shape leading to development of numerous cysts in the entire kidney.In this study, we analyzed the medaka mutant kintoun (ktu) that develops PKD. We revealed that renal epithelial cells of ktu mutant were flattened before beginning of urine flow. Cell transplantation experiments showed that ktu seemed to cause this morphological change in a cell-autonomous manner. To further understand the function of Ktu, we identified Ktu-interacting proteins and analyzed the roles of these proteins, especially in the developmental events requiring motile cilia in zebrafish.