Ectopic thyroid gland is rarely seen in the submandibular region, thereby posing difficult diagnostic and management problems. Two Caucasian women presented with painful swelling in the submandibular region, which increased in size considerably during the preceding months. Ultrasound-guided fine needle biopsy (Ug-FNB) revealed ectopic thyroid tissue. In conclusion, ectopic thyroid gland with or without pathology should be included in the differential diagnosis of submandibular swelling and Ug-FNB should be one of the first diagnostic tools utilized for this rare clinical entity. (J Oral Sci 53, 249-252, 2011)