An ectopic thyroid gland may be present in any location along the path of migration from the foramen caecum to the mediastinum. The most common locations for an ectopic thyroid are the lingual thyroid followed by median cervical cysts. An ectopic thyroid in the submandibular region is extremely rare. We present the case of a 44-year-old patient with ectopic thyroid tissue in submandibular space and a review of the literature related to it.