A 72-year-old female presented with a non-tender submandibular mass. The lesion had been present about 20 years with a recent rapid increase in size. Physical examination revealed a 5*5 cm-sized mass in the right submandibular region. Right submandibular gland resection was performed. The final histologic diagnosis was papillary thyroid carcinoma and atypical pleomorphic adenoma. Three years after operation she revisited with recurrent non-tender submandibular mass. Physical examination revealed a 6*7 cm-sized mass in the right submandibular region. Contrast-enhancement computerized tomography (CT) scans confirmed a 6*7-cm sized heterogeneously well enhancing mass similar to previous tumor. These finding suggested that recurrent submandibular tumor. Modified radical neck dissection of the right side was carried out, and the final diagnosis was carcinoma ex pleomorphic adenoma. The patient recovered well and discharged 15 days after surgery.