Lingual thyroid is an abnormal mass of ectopic thyroid tissue seen in base of tongue caused due to embryological aberrancy in development of thyroid gland. Most of the ectopic tissue is seen in the tongue. Their identification and proper management is essential since they may be the only functioning thyroid tissue occurring in the body. We report a case of lingual thyroid in a 40 year old female patient who was hypothyroid with posterior swelling of tongue. Tc-99 scintigraphy confirmed the clinical diagnosis and surgical excision of entire tissue was done by midline mandibular split osteotomy and patient was placed under lifelong thyroxine replacement. Follow up showed excellent results with minimum patient discomfort.