Introduction: Sclerosing mucoepidermoid carcinoma with eosinophilia is a rare morphological variant of thyroid carcinoma associated with Hashimoto's disease. To date, only three such tumors have been reported in the minor salivary glands. We describe the first case, to the best of our knowledge, of recurrent sclerosing mucoepidermoid carcinoma with eosinophilia in the minor salivary glands of the upper lip. Case presentation: A 61-year-old Japanese man was referred to our hospital with a mass in his median upper lip of four years' duration. An examination of his median upper lip revealed a well-defined tumor measuring 9×12mm in diameter, which was subsequently resected. Three years after the first surgery, the tumor recurred and was resected. Both tumors were confirmed by histopathology to be sclerosing mucoepidermoid carcinoma with eosinophilia. Neither recurrence nor metastasis was observed in three and a half years of follow-up after the second surgery. Conclusion: Our findings indicate that sclerosing mucoepidermoid carcinoma with eosinophilia can originate in the minor salivary glands and may be clinically or pathologically misdiagnosed as other conditions.
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