A 26-year-old woman, who was undergoing continuous ambulatory peritoneal dialysis, underwent a total parathyroidectomy for recurrent parathyroid adenoma. The surgeon did a total parathyroidectomy without autotransplantation. Hyperparathyroidism recurred. Tc99m-sestamibi parathyroid scintigraphy revealed focal increased activity on the submental region suggesting an ectopic parathyroid adenoma. Ultrasonographic evaluation showed several nodules with diameters of approximately 5 to 7 mm in the submental region. Unfortunately, this ectopic parathyroid lesion was not evaluated histopathologically because the patient refused reoperation. Her parathormone levels were suppressed with calcitriol. Because parathyroid adenomas are generally resistant to 1,25-dihydroxycholecalciferol, this parathyroid lesion appeared to be nodular hyperplasia rather than an ectopic parathyroid adenoma.