A 46-year-old man who has a child from a previous marriage without artificial reproductive technologies was referred to our hospital with a chief complaint of infertility. He had suffered from bilateral orchitis after parotitis six years ago. On physical examination, both testes were soft and 4 ml in size. Semen analysis showed azoospermia and the serum follicle stimulating hormone value was high (36.9 mIU/ml). Microdissection testicular sperm extraction was performed, and motile sperm were successfully retrieved. The histological examination showed increased thickness of the basement membrane and, peritubular fibrosis in most seminiferous tubules, with few focal areas of normal spermatogenesis.