We report a case of infertility due to obstructive azoospermia determi
ned by papillary cystadenoma of the epididymis. The 28-year-old patien
t had other signs of von Hippel-Lindau syndrome and died from neurolog
ical complications. The possibility of finding an epididymal neoplasm
should be kept in mind in any case of azoospermia with epididymal enla
rgement.