Twice-recurrent primary hyperparathyroidism due to parathyroid hyperplasia in an ectopic supernumerary gland

To describe a patient with multiple recurrences of primary hyperparathyroidism during a 24-year period.

We present the long-term history, including clinical and laboratory evaluations as well as findings on surgical interventions, in a 42-year-old man with recurrent hyperparathyroidism. In addition, the relevant literature is briefly reviewed.

At initial surgical exploration when the patient was 18 years old, a single enlarged parathyroid gland and a normal-appearing ipsilateral gland were found. After more than a decade without symptoms, the patient experienced recurrent symptomatic hypercalcemia. Surgical exploration revealed symmetric multiglandular disease that was treated by resection of 3 enlarged parathyroid glands and implantation of a portion of 1 gland in the forearm. After another 6-year asymptomatic period, the patient had recurrent symptoms and was found to have recurrent hypercalcemia. Surgical intervention revealed an enlarged supernumerary gland in an ectopic location. A preoperative technetium Tc 99m sestamibi scan and intravenous administration of methylene blue were helpful in identifying the ectopic parathyroid gland in the left carotid sheath.

This case illustrates the difficulty of determining which of a patient's parathyroid glands may become hyperfunctioning and the importance of considering whether supernumerary glands may be present. The case also demonstrates the need for long-term follow-up before a patient is considered "cured" of hyperparathyroidism.