Scrotal leiomyoma is a benign tumor derived from the scrotal dartos muscle; it is rare in clinical findings. The cause of the disease is not yet clear. Here, we present the case of a patient with no typical clinical features, but with some typical pathological changes. A middle-aged male patient saw a doctor for paroxysmal itching and burning of the scrotum. The physical examination revealed 3 × 5-cm(2), ill-defined, pink plaques, an uneven surface, and basal infiltration with pigmentation defects in the center. Pathology results showed dermal spindle cells in nodular hyperplasia. The nucleus was round at both ends, a perinuclear halo could be seen, and staining was positive for actin and negative for S100. Therefore, the diagnosis of scrotal myoma was made.