We report a case of a 66-year-old man with a simultaneous leiomyoma and contralateral smooth muscle hyperplasia of the epididymis. The left nodule showed characteristics of a benign leiomyoma consisting in the homogeneous proliferation of smooth muscle with a typical pattern of interlacing fascicles of spindled smooth muscle cells showing no mitotic activity. The right nodule was made up of bundles of smooth muscle, growing in a perivascular and interstitial pattern, without the homogeneous aspect of contralateral leiomyomatous tumours. The latter finding suggested non-neoplastic hyperplasia of the smooth muscle fibres of the epididymis. At 6 months after surgery, ultrasound of the scrotum showed no evidence of recurrent lesions.