Infantile myofibromatosis is a rare disease characterized by myofibroblastic proliferation, and typically occurs in early infancy. There is a wide spectrum of clinical presentation, which may involve various kinds of tissues in the body. Skin and subcutaneous lesions were the types of tissue most often seen. Although a multicentric form may behave aggressively, a solitary form of the tumor is benign with the possibility of spontaneous regression. Conservative management is justified after proper pathological diagnosis if the tumor involves an aesthetically important area. A case of solitary infantile myofibromatosis involving the upper lip is presented. Partial excision for biopsy was performed and long-term observation was undertaken. The tumor disappeared 3 years postoperatively.
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