Background: Ectopic thyroid tissue very rarely occurs in the buccal region. Even more rarely encountered is the papillary carcinoma arising in the ectopic tissue. Methods and Results: We herein present a 78-year-old female patient admitted with a giant, buccal mass that developed within the previous 2 years. Physical examination revealed a vegetative mass on the left buccal region without any cervical lymphadenopathy. Magnetic resonance imaging scan showed an 8- × 6-cm solid mass with cystic components in the left buccal region and thoracic computed tomography scanning revealed pulmonary metastasis. A successful surgery including lower lip splitting was performed, and the mass was totally excised with its surrounding capsule. Histopathologic examination revealed follicular variant of papillary carcinoma of the ectopic thyroid tissue. Conclusions: To our knowledge, our case is the first case of papillary carcinoma arising from ectopic thyroid tissue in the buccal region in the literature. © 2012 Wiley Periodicals, Inc.