Primary cilia are involved in various physiological regulations. We found that trichoplein, originally identified as a keratin-binding protein, suppresses the formation of primary cilia through binding to Aurora A kinase located at the ciliary base and regulates cell proliferation and differentiation. We also revealed that primary cilia of adipose progenitor cells are elongated in trichoplein knockout mice and that differentiation of adipose progenitor cells to adipocytes is suppressed in trichoplein knockout mice fed with a high-fat diet compared to those in the wild-type mice. It has been known that ectopic adipogenesis derived from fibro-adipogenic progenitor cells harms muscle regeneration after injury. Therefore, we have analyzed the role of trichoplein in tissue regeneration using trichoplein knockout mice and zebrafish. In this symposium, we would like to demonstrate the novel role of trichoplein in tissue regeneration with the regulation of primary cilia and discuss the possibility of trichoplein as a new therapeutic target.