Maternal virilization in pregnancy is associated, in most benign cases, with luteoma of pregnancy and hyperreactio luteinalis. Only a few reports relate this phenomenon to hyperthecosis or polycystic ovarian syndrome (PCOS), A case of recurrent maternal virilization during two consecutive pregnancies in a patient with PCOS is presented. In both pregnancies, the deepening of her voice, facial hirsutism and scalp hair loss began at the end of the first trimester and regressed 3-4 months post-partum. The patient underwent ovarian venous catheterization, and androgen secretion from both ovaries was found to be markedly high but similar, therefore ruling out an ovarian androgen-secreting tumour, Reviewing the English literature of similar cases, we found reports of only seven cases of maternal virilization during pregnancy associated with PCOS. Here, we present a case of recurrent maternal virilization in pregnancy associated with PCOS.