While mediastinal parathyroid carcinoma is a rare entity by itself, multiglandular coexistence of parathyroid carcinoma and parathyroid adenoma represents an extremely rare condition. Herein, we report such a rare presentation of multiglandular parathyroid neoplasm with an ectopic parathyroid carcinoma in the mediastinum and parathyroid adenoma on neck in a patient with persistent primary hyperparathyroidism (PHP). This 38-year-old female patient who initially presented with nephrolithiasis and osteopenia was subsequently diagnosed as having PHP. Following bilateral surgical neck exploration, existence of a parathyroid adenoma on the right side of neck was revealed. However, due to the persistence of hypercalcemia postoperatively, a single photon emission computed tomographic (SPECT/CT) examination was performed which showed a mass lesion in the anterior mediastinum. A surgical procedure was performed and the lesion, 10x10x3 mm in dimensions, confirmed to be a parathyroid carcinoma by histopathologic examination. At 1-year of follow-up there was no tumor recurrence or hypercalcemia. This is the first patient who had coexistent of ectopic mediastinal parathyroid carcinoma and parathyroid adenoma in the neck with persistent PHP. This case emphasises the difficulties in diagnosing and managing parathyroid disease. Multiple diagnostic studies are crucial for identification of multiple parathyroid glands in patients with persistent PHP.