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The case of a patient with primary hyperparathyroidism with the incidental finding of left thyroid hemiagenesis with isthmus involvement is presented. Thyroid hemiagenesis is a rare congenital abnormality. There have been approximately 310 cases reported in the literature to date. It has been reported as an incidental finding with a wide range of associated pathological conditions. It is important to consider this in patients being prepared for thyroid lobectomy due to the inevitability of postoperative hypothyroidsm. Due to the female predominance of thyroid disease, hemiagenesis was first thought to be more common in women. Left lobe cases accounts for the majority of instances and isthmus is absent in half the cases. Ultrasonography is the diagnostic tool of choice.