Anaplastic large cell lymphoma (ALCL) was recently recognized as a distinct type of non-Hodgkin's lymphoma characterized by anaplastic, large cells and expression of CD30 on immunohistochemical analysis. Since oral lesions of ALCL are rare, this tumor is not well documented in the literature. We report a case of ALCL of the maxillary sinus and back in a 79-year-old man who had an extensive swelling with an ulcer, extending from the left cheek to the upper gingiva, and a subcutaneous mass of the back. Computed tomography and magnetic resonance imaging of the gingival swelling revealed a bulky tumor in the left maxillary sinus with bone destruction. Histological examinations of both lesions were performed. The lesions were both diagnosed as CD30-positive, null cell type ALCLs. Complete remission was achieved by six courses of CHOP chemotherapy. There have been no signs of recurrence 10 months after treatment; however, long-term follow-up is necessary because of a relatively high rate of recurrence.