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BACKGROUND: Polycystin was first discovered in autosomal dominant polycystic kidney disease, which was involved in the formation of cysts, and later it was found to be related to bone formation and the occurrence and development of bone-related diseases such as craniosynostosis, bone loss, and osteosarcoma. OBJECTIVE: To review the role of polycystin in bone formation mechanism and the occurrence and development of bone-related diseases. METHODS: PubMed database was searched for relevant documents from January 1990 to April 2020. The search terms were “bone” or “bone formation mechanism” or “craniosynostosis” or “bone loss” or “osteosarcoma” and “polycystin.” At the same time, CNKI database was searched for relevant documents from January 1979 to April 2020. The search terms were “polycystin” and “bone,” “polycystin” and “bone formation mechanism,” “polycystin” and “bone loss,” “polycystin” and “osteosarcoma,” “polycystin” and “craniosynostosis.” Finally, 45 relevant documents were included for review. RESULTS AND CONCLUSION: Polycystin exists as a mechanoreceptor in the plasma membrane of bone cells, which is activated by external mechanical stimuli, and triggers signaling pathways in the cells, participates in the regulation of bone formation and bone loss mechanisms, participates in bone development, and plays an important role in the occurrence and development of bone diseases, such as craniosynostosis, bone loss, and osteosarcoma. To date, there have been few studies regarding the role of polycystin in maxillofacial bone and skull diseases, suggesting that in-depth studies on the role and mechanism of polycystin in the physiopathological processes of the skeletal bone are warranted. [ABSTRACT FROM AUTHOR]