Two cases of desmoplastic form of ameloblastoma are reported. This tumor shows marked stromal desmoplasia and often scattered osteoplasia. The majority of stromal elements is represented by myofibroblasts. The epithelial component of this tumor is often scanty and different from epithelial islands of classic ameloblastoma. Pathological, immunohistochemical and radiological findings are described and the differential diagnosis of this tumor is discussed.