Congenital lipomatosis of the face is characterized by collections of nonencapsulated, mature lipocytes which infiltrate local tissues and tend to recur after surgery. These lesions represent a distinct clinicopathologic entity that has not been previously reported in this location in children. Three children with congenital lipomatosis of the face were treated at the Institute of Reconstructive Plastic Surgery over a 2- to 14-year follow-up period. In each instance, pathologic evaluation by light and electron microscopy revealed similar lesions sharing the following morphologic criteria: (1) nonencapsulated tumors containing mature fat cells, (2) infiltration of adjacent muscle and soft tissue, (3) absence of malignant characteristics, (4) absence of lipoblasts, (5) presence of fibrous elements in conjunction with increased numbers of nerve bundles and vessels, and (6) hypertrophy of subjacent bone. All three lesions recurred after numerous excisions, some of which were extensive. All were benign by histologic examination and remained so for as long as 14 years. Surgical treatment improved the aesthetic appearance of each child despite evidence of tumor persistence. Although these tumors are benign, we recommend an early aggressive surgical approach to control the infiltrative nature of their growth and to improve facial appearance.