Imaging technologies in the differential diagnosis and follow-up of brown tumor in primary hyperparathyroidism: Case report and review of the literature

Diacinti, Davide; Cipriani, Cristiana; Biamonte, Federica; Pepe, Jessica; Colangelo, Luciano; Kripa, Endi; Iannacone, Antonio; Orlandi, Martina; Guarnieri, Vito; Minisola, Salvatore

doi:10.1016/j.bonr.2020.100745

Cited by 8 publications

(4 citation statements)

References 35 publications

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“…Radiographically, BT typically present as well-de ned lytic lesions, sometimes showing intralesional trabeculations and cortical bone thinning (10). Signs of HPT may be expressed alongside other indicators such as the loss of the lamina dura and the classic "salt and pepper" appearance of the skull which were evident in this case.…”

Section: Discussionmentioning

confidence: 65%

Tooth mobility revealing a brown tumor associated with normocalcemic tertiary hyperparathyroidism: case report and literature review

Boukssim,

Damiri,

Chami

2024

Preprint

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Background Brown tumors are rare non neoplastic lesions resulting from abnormal bone metabolism in hyperparathyroidism. They are rarely observed in tertiary hyperparathyroidism. Tertiary hyperparathyroidism is a very rare condition that arises in the setting of renal disease. It is the autonomous secretion of parathyroid hormone leading to persistent hypercalcemia and elevated serum parathormone despite resolution of the underlying problem. Case presentation: we report an unusual case of tooth mobility revealing the presence of a brown tumor located in the mandible in a north African 41 year old male with chronic kidney disease and a history of parathyroidectomy. Clinical examination revealed firm vestibular swelling. Radiographic assessment showed a well-defined periapical radiolucent lesion, consistent with a giant cell lesion. Elevated parathyroid hormone levels (3283.0 pg/ml), normocalcemia, and hyperphosphatemia supported the diagnosis of a brown tumor due to tertiary hyperparathyroidism. The treatment of brown tumor consists of addressing the underlying hyperparathyroidism. The unusual observation is that the patient presented with elevated parathormone level associated with normocalcemia raising the question on the indication for parathyroidectomy in this case. Conclusion The patient initially benefited from a parathyroidectomy with a reduction in serum parathormone by over 50% and tumor regression. However, 2 months later, we observed a recurrence of parathormone levels ,while calcium levels remained normal, necessitating the use of high dose calcimemitic agents. This case highlights the importance of thorough evaluation in patients with chronic kidney disease and persistent hyperparathyroidism, and also the question of the indication of parathyroidectomy in normocalcemic tertiary hyperparathyroidism

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Section: Discussionmentioning

confidence: 65%

Tooth mobility revealing a brown tumor associated with normocalcemic tertiary hyperparathyroidism: case report and literature review

Boukssim,

Damiri,

Chami

2024

Preprint

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“…HPT-JT syndrome is a rare autosomal dominant disorder characterised by the occurrence of hyperparathyroidism at an early age with a high incidence of parathyroid carcinomas, atypical adenomas, and ossifying fibromas of the jaw. In addition, in some rare cases, brown tumors may occur elsewhere [ 9 ], and there may be the presence of renal lesions and uterine tumors [ 10 ]. According to the literature, germline inactivating mutations of the CDC73 gene have been observed in approximately 50–75% of HPT-JT cases and 14% of FIHP [ 11 ].…”

Section: Discussionmentioning

confidence: 99%

Brown tumor due to primary hyperparathyroidism in a familial case: a case report

Diao,

Zhang,

Zhao

et al. 2023

BMC Endocr Disord

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Background Primary hyperparathyroidism (PHPT) is an uncommon disorder characterised by hypercalcemia with an increased parathyroid hormone level. We reported a PHPT familial case with two subjects, a father and a daughter, and both of them had suffered from the brown tumor. Case presentation The proband, a 43-year-old patient, developed parathyroid adenomas at the age of 15; a histologically confirmed right parathyroid adenoma was removed by parathyroidectomy; and after six months follow-up, the serum calcium level was normalised. At the age of thirty-three, a CT scan of his head and neck revealed a mass in the right maxilla, as well as PHPT (i.e., left inferior parathyroid adenoma). Then, he underwent a biopsy of an exophytic lesion in the right maxilla and was diagnosed by pathology as a brown tumor, with the serum calcium and PTH levels at 2.78 mmol/L and 221 pg/mL, respectively. Subsequently, the patient took a left inferior parathyroid microwave ablation with ultrasound guidance. After three months of follow-up, the serum calcium and PTH levels returned to normal, and the brown tumor was resolved. After three years, it mineralised as revealed in a CT scan. By the time he was 43 years old, during the 28-year follow-up period, the serum calcium and PTH levels were still within the normal range, and there was no discomfort reported. He has consistently taken calcium supplements throughout the 28 years. Since the initial diagnosis, his blood indicators of kidney function have been normal, and ultrasound showed renal calculus in the right kidney and a normal left kidney. The proband’s daughter, a 15-year-old girl, experienced left upper extremity pain for ten months. CT scan revealed a mass in the distal left radius, and a giant cell tumor was suspected. A surgical internal fixation was performed, and the pathology showed a brown tumor. Laboratory tests revealed a serum parathyroid hormone (PTH) level of 1554pg/mL, calcium level of 3.14 mmol/L, phosphorus level of 0.72 mmol/L, and alkaline phosphatase level of 1892 U/L. Given the osteitic changes and elevated levels of calcium and PTH, ultrasonography was performed, after which a mass was detected measuring 19 × 9 × 7 mm mixed with solid components and cystic fluid in the right thyroid gland. The results of 99mTc-MIBI scintigraphy confirmed the abnormal accumulation of 99mTc-MIBI in the right thyroid gland but not seen in the bilateral parathyroid glands. The patient underwent thyroidectomy, and the postoperative pathology report indicated an intra-thyroid ectopic parathyroid adenoma. The serum calcium and PTH levels became normal at 4 h after surgery. One to three months after operation, the serum calcium level was low, while the serum PTH level was high. Then, the patient was advised to take calcium supplements. Until the sixth month after the operation, the serum calcium level and serum PTH level returned to normal, and the bone pain was relieved. The patient’s blood tests for kidney function remained normal. There was no evidence of bilateral kidney disease (such as nephrolithiasis or nephrocalcinosis) detected by ultrasound scan. There were several similarities in the state of illness between these two subjects. Both the father and the daughter developed parathyroid adenomas at the age of 15, and there was no lesion in other endocrine glands. And genetic testing revealed mutations in the CDC73 genes in both father and daughter. On the other hand, there were also a few differences. The father’s first signs of brown tumor were in the right maxilla, while the daughter’s appeared in the distal left radius. The father presented pathological changes in the left and right parathyroid glands, whereas the daughter presented with an ectopic parathyroid adenoma in the right thyroid gland. Conclusion We report a familial case in which father and daughter were diagnosed to have brown tumors due to parathyroid adenoma and ectopic parathyroid adenoma, and genetic testing revealed CDC73 gene mutations in both. Therefore, in the diagnostic and differential process of young patients having bone disease, clinicians should not only focus on the clinical manifestations of the skeleton, but also implement a comprehensive analysis of systemic symptoms, considering the possibility that the patient has familial PHPT.

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“…In MR images, solid components have heterogenous signal intensity on T2-weighted images, whereas cystic components have hyperintensity on T2-weighted images (35). Based on the components, lesions of brown tumors can be classified into three types: solid lesions with low-iso intensity on T1 and T2 weighted images and contrast enhancement by gadolinium; mixed solid and cystic lesions with low-iso intensity on T1 weighted images and iso-high intensity on T2 weighted images, contrast enhancement of solid portions and the septa of cystic lesions; cystic lesions with high intensity on T2 weighted images and contrast enhancement of the periphery and septa of the lesions (39). Because of the multiple osteolytic lesions, brown tumors sometimes mimic metastatic carcinoma, bone cysts, osteosarcoma, and giant cell tumors of the bone (15,20,22,36,(40)(41)(42)(43).…”

Section: Discussionmentioning

confidence: 99%

Multiple Bone Cysts Caused by Hyperparathyroidism: A Case Report and Review of the Literature

MIWA,

TANAKA,

AIBA

et al. 2023

CDP

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Background: Brown tumor, a skeletal complication of severe hyperparathyroidism, comprises reparative granulation tissue and proliferating fibrous tissue with hemosiderin deposition. Multiple brown tumors are extremely rare complications of primary hyperparathyroidism. Case Report: A 41-year-old woman presented with pain in the left knee. Radiography showed multiple cystic lesions in both femurs and the left proximal tibia, and additional radiography showed multiple cystic lesions in the left humerus and ulna. Magnetic resonance imaging (MRI) revealed multiple cystic lesions in the bilateral femurs, left proximal tibia, and ilium. Laboratory tests revealed hypercalcemia (albumin-corrected calcium level, 13.9 mg/dl), hypophosphatemia (phosphate level, 1.6 mg/dl), elevated level of alkaline phosphatase level (614 U/l), and markedly elevated parathyroid hormone (PTH) level (1,070 pg/ml; normal range=10-65 ng/l). 99mTc-hexakis-2-methoxyisobutyl-isonitrile scintigraphy revealed tracer accumulation in the left upper parathyroid gland, which was consistent with parathyroid tumor. Although resection of the parathyroid tumor was planned, the patient developed parathyroid apoplexy before tumor excision. After the parathyroid apoplexy, serum calcium and PTH levels temporarily normalized. Resurgence of the PTH level was observed 2 years after the diagnosis, and the patient underwent left upper parathyroidectomy. One year after the tumor excision, the patient had no symptoms, and MRI showed shrinkage of the cystic bone lesions. Conclusion: This report emphasizes the importance of considering hyperparathy-roidism as a differential diagnosis for patients with multiple bone lesions.

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Imaging technologies in the differential diagnosis and follow-up of brown tumor in primary hyperparathyroidism: Case report and review of the literature

Cited by 8 publications

References 35 publications

Tooth mobility revealing a brown tumor associated with normocalcemic tertiary hyperparathyroidism: case report and literature review

Tooth mobility revealing a brown tumor associated with normocalcemic tertiary hyperparathyroidism: case report and literature review

Brown tumor due to primary hyperparathyroidism in a familial case: a case report

Multiple Bone Cysts Caused by Hyperparathyroidism: A Case Report and Review of the Literature

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