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Primary squamous cell carcinoma (PSCC) is a rare neoplasm of the thyroid with a very poor prognosis. We report a case of a 42-year-old woman with occasionally found mass in the right anterior area of the neck. After a total thyroidectomy, histopathology and immunohistochemistry tests confirmed primary squamous cell carcinoma of the thyroid with the exclusion of all other possible primary tumor locations. 5 months later, PET scan discovered abnormality in right cervical lymph nodes with a fine needle aspiration confirming to be tumor recurrence. After a modified radical neck dissection was performed with pathological results of the neoplasms being PSCC of the thyroid origin, a full course consecutive radiotherapy was then followed. Due to a prompt diagnosis and the complete dissection of primary tumor and metastatic lymph nodes, no recurrence was observed at the follow-up visits. Comparing to the published cases of PSCC of the thyroid, our paper stated a whole process of diagnosis and standardized treatment, together with classical matched figures of pre-op examinations and dissected specimen. Furthermore, a review of the present literatures summarized the diagnosis, treatment and prognosis of thyroid PSCC. The management of PSCC requires a multi-disciplinary approach.