A Case of Primary Hyperparathyroidism with Brown Tumor of the Maxilla

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Brown tumors are thought to be a sequela of bone remodeling caused by persistent excessive levels of parathyroid hormone. It may occur in any bone in the body, and may manifest itself as multiple bone lesions mimicking bone metastasis of a malignant tumor. We herein report on a rare case of primary hyperparathyroidism associated with a brown tumor of the maxilla. A 52-year-old woman with a 6-month history of increasing cheek swelling, pain in the costal region, and epigastric pain was referred to our hospital. CT and MRI revealed a large osteolytic mass in her left maxilla protruding toward the nasal cavity and underneath the cheek. CT also showed a mass just behind the right thyroid lobe. On systemic bone scintigraphy, marked isotope uptake was observed in multiple bones. Under a tentative diagnosis of malignant maxillary tumor with multiple bone metastasis, open biopsy of the maxilla was performed via an anterior antrostomy. The histopathological finding did not indicate malignancy, but was consistent with brown tumor. Further systemic examinations were then carried out; Hypercalcemia and hyperparathyroidism were found on serological tests; multiple osteolytic lesions in the costae, cranium, and phalanges of the hand were seen on radiological examinations; and isotope uptake was identified behind the right thyroid lobe on 201Tl-Cl/99mTcO4−-subtraction scintigraphy. The patient was diagnosed as having a parathyroid tumor associated with primary hyperparathyroidism and brown tumor, and received intravenous bisphosphonate, which turned out to be ineffective. She eventually underwent resection of the parathyroid tumor. The histopathological diagnosis was parathyroid adenoma. Except for temporary hypocalcemia with tetany, her postoperative clinical course was uneventful. The bone lesions including the brown tumor of the maxilla gradually decreased thereafter. We should be aware that not only metastatic malignant tumors but also hyperparathyroidism may cause multiple osteolytic lesions. The clinical characteristics, diagnosis, and treatment of hyperparathyroidism with brown tumor were bibliographically reviewed.

A Case of Primary Hyperparathyroidism with Brown Tumor of the Maxilla

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