A 30-year-old man present with infertility for 2 years. Magnetic resonance imaging (MRI) revealed two right testes in the scrotum and inguinal region, respectively. Semen analysis revealed cryptozoospermia. Polyorchidism was considered to have caused spermatogenic dysfunction and male infertility. We performed right high orchiectomy and simultaneous testicular sperm extraction on the same testis. To our knowledge, this is the first case of testicular sperm extraction performed for patients with polyorchidism.