BORIS (brother of the regulator of imprinted sites) was first expressed in amniotes during the evolution of vertebrates. Under human physiological conditions, this protein is highly expressed in male testes only. The expression sequence of BORIS in male mice is highly consistent with the sequence of spermatogenesis. Male BORIS knockout mice are sterile, while females are normal. BORIS induces the expression of specific genes in germ cells and has an important role in spermatogenesis, specifically regulating spermatogenesis under normal physiological conditions. Referring to recent studies of BORIS transgenic mice, BORIS knockout mice, and BORIS expression in testis, this review summarizes the expression and localization of BORIS in testis, the mechanism of BORIS in spermatogenesis, and the functional research and drug screening of BORIS, providing a new reference for molecular targeted therapy.