A cross-sectional study of benign breast disease (BBD) was conducted to determine the actual prevalence and follow-up importance of BBD among women with polycystic ovary syndrome (PCOS) in conjunction with an ongoing prospective cohort study. The present study involved a subset of the original group of 244 women with a diagnosis of PCOS and 244 control women matched by age and race. A total of 240 women (116 cases and 124 controls) were included in the present analysis. The majority of women in each group were Caucasians (93 and 96%, respectively). The median age was 46 years in the cases and 47 years in the controls. Screening mammography begins at the age of 40 and has been carried out in 69% of cases and 66% of controls since the study began. Family history of breast disease was observed in 27 cases of both the groups (p > 0.05). Neither fibrocystic breast disease, lump thickening, calcification, fibroadenoma, pain, redness, discharge nor hyperplasia showed a significantly higher prevalence rate in cases than in controls. Eleven (9%) women with PCOS and 21 (17%) controls underwent diagnostic or curative surgery (relative risk: 0.56). These results, in contrast to the previously published literature, do not allow us to conclude that there is a higher risk for BBD among women with PCOS, and the proportion of women with a positive family history of breast cancer was significantly greater in women with PCOS compared with controls. Our observation is that having PCOS does not appear to affect surgeons' decisions to remove BBD.