A 24-year-old Caucasian man was evaluated because of a history of multiple firm, reddish, papulonodular skin lesions occurring over the buttocks, thighs, and lower portions of his back and legs since the age of 17 years. Most lesions had developed within 1 year of onset and had become very painful. The family history revealed that the patient's mother and maternal aunts had similar skin lesions. The mode of inheritance is that of autosomal dominant pattern. The mother and three maternal aunts all had had hysterectomies. Biopsy specimens of skin lesions with hematoxylin-eosin and Masson-trichrome stains showed typical leiomyoma. Bodian's staining method revealed an increased amount of nerve fibers interlacing within muscle fibers and in the surrounding tissue. This finding may explain the painful nature of the cutaneous leiomyomas. Treatments consist of analgesics for control of pain and selective excision of larger painful leiomyomas.