Cutaneous piloleiomyoma, angioleiomyoma and genital leiomyoma are variants of superficial cutaneous leiomyoma. The main purpose of this paper was to present clinical, histopathological and immunohistochemical diagnosis criteria for an unusual case of pilar leiomyoma in an 18-year-old male patient. The initial clinical aspect was very similar to spontaneous eruptive keloids: red-violet color, painless, aspect of "crab pincers" of some of the lesions, epidermis atrophy, telangiectasia, located on acneiform zones and compliance with cephalic extremity. The patient had no history of trauma, surgery or acne. Local treatment of one lesion was performed with cryotherapy using liquid nitrogen (-172°C) together with intra-lesion steroid injections, occlusive dressings and silicone gel. Local therapy did not showed notable results, moreover the lesion become painful. Skin biopsy with histological and immunohistochemical analysis revealed the diagnosis of multiple cutaneous pilar leiomyoma without atypia. The particularity of the case stands in the atypical onset followed by explosive increasing of lesions number and the appearance of pathognomonic pain after local therapy.