Normocalcemic hyperparathyroidism presented with mandibular brown tumor: report of a case
Introduction
In 1891, Von Recklinghausen recognized the clinical impact of hyperparathyroidism and described osteitis fibrosa cystica as the pathognomonic bone lesions of this entity. In 1926, Mandl operated a parathyroid tumor in a patient with hypercalcemia and radiological changes of osteitis fibrosa cystica and demonstrated postoperative regression of the bone disease and biochemical abnormalities [1]. Incidence of primary hyperparathyridism has been reported as approximately five per 10000 population per year. Among patients aged more than 60 years, the frequency is even higher, approaching nearly one per 1000 in men and twice that in women [2]. Osteitis fibrosa cystica is classically associated with primary hyperparathyroidism. However, it is an unusual manifestation with involvement in less than 2% of primary hyperparathyroidism cases [3].
The term Brown tumor is derived from the characteristic macroscopic appearance of brownish material within the cystic lesion. This appearance results of blood pigments that are both free and within hemosiderin-laden macrophages. These “tumors” are hypervascular and intensely active on bone scans [4]. Since this lesion is reactive rather than neoplastic, the Brown tumor is not a tumor at all [5]. Although Brown tumor is very unusual entity in ENT practice, it may encountered in head and neck, in which mandible is the most common location and is involved in 4% of patients with osteitis fibrosa cystica [6]. Brown tumor has also been reported in the paranasal sinuses [7].
Brown tumor most commonly presents as a slowly enlarging, painful mass. That can be locally aggressive, without metastatic potential [8]. When present 90% of osteitis fibrosa cystica occurrences in hyperparathyroidism are due to parathyroid adenoma, of the remaining 10% are due to primary hyperplasia of parathyroid gland. We report an unusual case of Brown tumor in the mandible associated with a normal serum calcium level.
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Case report
A 62-year-old female patient had been presented with a mass on her mandible for 7 months. Symptoms of the patient appeared 3 years ago as pain on her left leg and walking difficulties. One year later, she recognized a swelling on her left leg. She was admitted to a hospital for further investigation of left midtibial mass (Fig. 1). The bony lesion had been excised and reconstructed by a bone graft. The histopathologic diagnosis was Brown tumor. The patient had no other abnormality on her
Discussion
Early diagnoses and successful treatment regimes of hyperparathyroidism have made clinical evidence of bone disease uncommon. During the last three decades primary hyperparathyroidism has been recognized much more commonly because of the routine determination of serum calcium level by new methods and the objective parathyroid hormone radioimunoassay technique. Hyperparathyroidism is a well-known cause of hypercalcemia, and serum calcium level with musculoskeletal complications should have been
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Cited by (34)
A Diagnosis of Normocalcemic Primary Hyperparathyroidism Prompted by “Salt and Pepper” Lesions of the Calvarium
2022, AACE Clinical Case ReportsCitation Excerpt :Previous reports have described the “salt and pepper” appearance of the calvarium in cases of hypercalcemic primary hyperparathyroidism and secondary hyperparathyroidism but typically not in cases of normocalcemia.11-13 However, 1 case report described a case of mandibular brown tumor (osteitis fibrosa cystica) in a patient who was ultimately found to have normocalcemic hyperparathyroidism but notably in the setting of 25-hydroxyvitamin D deficiency and parathyroid adenoma.14 Although the patient in the abovementioned case report shares similarities with the patient presented here, the crucial difference is that secondary causes of hyperparathyroidism were excluded in the case of our patient.
Skeletal lesions in primary hyperparathyroidism
2015, American Journal of the Medical SciencesCitation Excerpt :Biochemical screening is also important. Serum calcium and PTH should be examined at the same time because normal serum calcium occurs often in PHPT.30,33 A variety of bone imaging methods in combination with parathyroid radiography are also helpful to make the diagnosis.
Leontiasis ossea: A presentation of hyperparathyroidism in an indigenous australian man secondary to chronic renal failure
2013, Journal of Oral and Maxillofacial SurgeryMaxillary brown tumor as initial presentation of parathyroid adenoma: A case report
2012, Kaohsiung Journal of Medical SciencesCitation Excerpt :The first choice in managing brown tumor caused by primary hyperparathyroidism is parathyroidectomy. Most of the bone lesions will regress with time after parathyroidectomy, thus, surgical removal of the brown tumor may not be necessary [2–5]. However, at least two studies have shown that the tumors failed to regress and became even larger after parathyroidectomy.
Normal ionized serum calcium is a prerequisite for characterization of normocalcemic primary hyperparathyroidism
2012, American Journal of the Medical SciencesNormocalcemic Parathyroid Adenoma with Brown's Tumor Maxilla: A Rare Case
2023, Iranian Journal of Otorhinolaryngology