Thyroglossal duct cysts are most common neck masses after benign lymphonodes. They originate from primitive thiroglossal duct, so they could be locate along its course. Every mass in the middle line of the neck can be considered as a thyroglossal cyst. Best treatment is surgery (Sistrunk procedure). We present a case of unusual localization at floor of the mouth of thyroglossal in a 34 years old woman. To our knowledge in literature, only two cases, have been reported both.
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