Introduction: Klinefelter syndrome (KS) is the most common sex chromosomal anomaly with hypogonadism being a common feature in this syndrome. The ejaculate volume is about normal to low in men with this syndrome. In the present report, we describe the successful treatment of a lifelong anejaculation in a man with KS. This represents the first report of successful treatment of anejaculation in KS. Case presentation: A 24-year-old Caucasian man presented with a lifelong history of failure of ejaculation. A diagnostic work-up revealed the existence of KS and administration of human chorionic gonadotrophin restored the ejaculation. Conclusion: This case report confirms further the increased prevalence of ejaculatory disorders among men with KS in whom associated hypogonadism should be considered a cause of anejaculation. Diagnosis of KS is often delayed. Early diagnosis of the syndrome before puberty is highly recommended and desirable to maintain quality of life.

Klinefelter syndrome, hypogonadism, anejaculation, human chorionic gonadotrophin