Patients with extragonadal seminoma are at high risk of developing a p
rimary testicular neoplasm many years after the initial diagnosis and
therapy. Therefore, long-term follow-up is critical in the proper mana
gement of these patients. We present the first case of Leydig cell hyp
erplasia, mimicking a testicular neoplasm, 21 years after diagnosis an
d treatment of extragonadal seminoma.