We report a rare case of primary hyperparathyroidism with a functioning parathyroid cyst in a 45-year-old male. He was a recurrent stone former, and consulted our hospital for further examinations of hypercalcemia. Plasma levels of intact parathyroid hormone (PTH) were elevated to 130 pg/ml. Ultrasonography, computed tomography and magnetic resonance imaging revealed a parathyroid cyst on the right lobe of the thyroid gland. We performed right superior parathyroidectomy. Histological examination demonstrated a secondary pseudocyst resulting from cystic degeneration of a parathyroid adenoma. Plasma levels of intact PTH normalized after operation. To our knowledge, only 48 cases of functioning parathyroid cyst were reported in the Japanese literature. We discuss the clinical features and histological evidence of functioning parathyroid cyst.
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