Anti-epileptic drugs may have negative effects on sexual function and hormonal profile. The exact mechanisms involved, however, have yet to be completely understood. We report a case of ejaculation failure and abnormal hormonal profile in a patient affected by epilepsy. A 59-year-old man, under treatment with valproate and phenytoin for 15 years, complained of orgasmic an ejaculation over the previous 6 months. He was not affected by other relevant pathologies and he had not undergone pelvic surgery. We found spermatozoa in post-orgasmic urine, which confirmed our suspicion of retrograde ejaculation. The hormonal profile showed high levels of FSH, LH and, surprisingly, increased levels of total testosterone and SHBG. We hypothesized bladder sphincter inhibition and receptor alterations due to the anti-epileptic drugs.