Overrepresentation of polycystic ovary syndrome (PCOS) in women with epilepsy has been described since the early 1980s. While some authors attribute this association to an effect of the seizure disorder on the hypothalamic control of reproductive function, others have reported a relationship with the use of the antiepileptic drug valproic acid (VPA). In this article we review the literature on this complex issue, with a detailed analysis of the different reports which describe the reproductive endocrine assessment in women with epilepsy. In spite of the large number of patients assessed, a clear picture does not emerge, mostly because of the wide variability of methodology employed in the different study projects and of the small size of many patient samples especially when divided in subgroups. However, on the whole these studies suggest that women with epilepsy are at risk for developing reproductive endocrine disorders, even if there is not yet definite evidence that PCOS may be over-represented in these patients nor that VPA may be the cause of endocrine problems. It is likely that both the epileptic disorder and the antiepileptic treatment play different roles in the development of such disturbances. This hypothesis deserves further prospective study in large samples of patients; consistency in methodology, diagnostic criteria and presentation of results should always be encouraged in the researchers dealing with these projects. In the meantime, women with epilepsy should be carefully monitored with regard to menstrual function, bodyweight and hyperandrogenism, and evaluation of these parameters should become part of the routine evaluation in baseline and follow-up consultations.