Congenital dilatation of the Stensen's duct is an extremely rare disease, which may have a hereditary background. However, there are no reports about the familial occurrence of congenital Stensen's duct dilatation. Herein, we present the first case of congenital dilatation of Stensen's duct in siblings. Therefore, we suggest that congenital dilatation of the Stensen's duct is an extremely rare hereditary disorder.