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Thyroid hemiagenesis (THG) is a rare congenital anomaly in which one lobe of thyroid gland fails to develop. Agenesis may be unilateral, total or isthmic. Left thyroid lobe is commonly involved than right lobe in hemiagenesis. Clinically patients can be euthyroid, hypothyroid or hyperthyroid. Often it is diagnosed as an incidental finding during ultrasonography (USG) study of neck, which easily diagnose this condition. Actual incidence of THG is unknown, most cases are diagnosed in patients admitted for thyroid scan or thyroid surgery because of suspicion of other thyroid abnormalities. This explains high frequency of association of hemiagenesis with other thyroid abnormalities such as multinodular goiter, adenoma, hyperthyroidism, hypothyroidism, chronic thyroiditis, and carcinoma. We report a case of 58-year-old male patient detected to have hemiagenesis of left thyroid lobe and isthmus when USG neck was performed for carcinoma right buccal mucosa with metastatic cervical lymphadenopathy.