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Branchial cyst is the most common cyst to arise in the lateral neck, and frequently appears in the second or third decades. However, ectopic thyroid tissue within this cyst is a rare abnormality, and papillary carcinoma arising in this ectopic tissue is extremely rare. According to our knowledge, only three cases of this disease have been reported in English literature previously. A 34-year-old woman with papillary carcinoma arising in ectopic thyroid tissue within a branchial cyst was presented in this paper.