Brown tumor of the jaw as a rare manifestation of hyperparathyroidism: Two case reports and literature review

Hakkou, F.; Benjelloun, L.; Hallab, L.; Chbicheb, S.

doi:10.1016/j.ijscr.2023.108823

Cited by 3 publications

(2 citation statements)

References 10 publications

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“…Excessive production of PTH 4 - 10 may occur due to primary hyperparathyroidism (PHP), phosphate retention, skeletal resistance to PTH, impaired PTH degradation, and altered calcium-PTH feedback regulation in secondary hyperparathyroidism (SHP) or persistent tertiary hyperparathyroidism (THP) 2 , 3 , 11 , 12 . Increased PTH production results in hypercalcemia 3 , due to increased calcium absorption in the intestine, increased renal tubular reabsorption, and increased osteoclastic activity 4 , 5 , 11 which leads to bone demineralization, resulting in microfractures hemorrhage, hemosiderin deposition 13 , and excessive vascular proliferation that give such lesions the characteristic brown staining, justifying the nomenclature brown tumor of hyperparathyroidism (BTH) 4 , 5 .…”

Section: Introductionmentioning

confidence: 99%

“…Brown tumor of hyperparathyroidism secondary to PHP is very rare 6 , 15 —only 2–5% of its carriers have this condition, usually caused by massive PTH secretion 6 , 12 , 16 , 17 . PHP can occur due to parathyroid adenoma 4 , 5 , 10 , 13 , 16 (up to 85% of cases) 4 , 5 , 10 —benign but metabolically active 4 , 5 , eventually ectopic lesion 7 ; parathyroid carcinoma 4 , 5 , 10 —which, although a rare cause of PHP (<1% of cases), presents bone involvement (BTH) more frequently (up to 90% of cases) when compared with benign causes of PHP 4 , 5 ; and hereditary factors (5–10% of cases) such as MEN type 1 (comprises up to 95% of hereditary cases of BTH) and 2A, HP-jaw tumor syndrome, and familial isolated HP that can result in BTH if undiagnosed 2 , 4 .…”

Section: Introductionmentioning

confidence: 99%

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Update on brown tumor of hyperparathyroidism

Guedes,

Becker,

Nakagawa

et al. 2024

Rev. Assoc. Med. Bras.

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Section: Introductionmentioning

confidence: 99%

Section: Introductionmentioning

confidence: 99%

Update on brown tumor of hyperparathyroidism

Guedes,

Becker,

Nakagawa

et al. 2024

Rev. Assoc. Med. Bras.

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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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Brown Tumors: The Hidden Face of Primary and Renal Hyperparathyroidism Amid Real-Life Settings

Carsote,

Ciobica,

Sima

et al. 2024

JCM

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Brown tumors, an exceptional bone complication of severe primary (PHP) or renal (secondary) hyperparathyroidism (RHP), are caused by long-standing, elevated parathormone (PTH)-induced osteoclast activation causing multinucleated giant cell conglomerates with hemosiderin deposits in addition to the local production of cytokines and growth factors. We aim to present an adult case series including two females displaying this complication as part of a multidisciplinary complex panel in high PTH-related ailments. The approach was different since they had distinct medical backgrounds and posed a wide area of challenges amid real-life settings, namely, a 38-year-old lady with PHP and long-term uncontrolled hypercalcemia (with a history of pregnancy-associated PHP, the removal of a cystic jaw tumor, as well as a family and personal positive diagnosis of polycystic kidney disease, probably a PHP-jaw tumor syndrome), as well as, a 26-year-old woman with congenital single kidney and chronic renal disease-associated RHP who was poorly controlled under dialysis and developed severe anemia and episodes of metabolic acidosis (including one presentation that required emergency hemodialysis and was complicated with convulsive seizures, followed by resuscitated respiratory arrest). Both subjects displayed a severe picture of PHP/RHP with PTH levels of >1000 pg/mL and >2000 pg/mL and elevated serum bone turnover markers. Additionally, they had multiple brown tumors at the level of the ribs and pelvis (asymptomatically) and the spine, skull, and pelvis (complicated with a spontaneous cervical fracture). As an endocrine approach, the control of the underlying parathyroid disease was provided via surgery in PHP (for the postparathyroidectomy hungry bone syndrome) via medical intervention (with vitamin D analogs) in RHP. Additionally, in this case, since the diagnosis was not clear, a multidisciplinary decision to perform a biopsy was taken (which proved inconclusive), and the resection of the skull tumor to confirm the histological traits. This series highlights the importance of addressing the entire multidisciplinary panel of co-morbidities for a better outcome in patients with PHP/RHP-related brown tumors. However, in the instance of real-life medicine, poor compliance and reduced adherence to recommendations might impair the overall health status. Thus, sometimes, a direct approach at the level of cystic lesion is taken into consideration; this stands for a narrow frame of decision, and it is a matter of personalized decision. As seen here, brown tumors represent the hidden face of PHP/RHP, primarily the complex and severe forms, and awareness is essential even in the modern era.

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Brown tumor of the jaw as a rare manifestation of hyperparathyroidism: Two case reports and literature review

Cited by 3 publications

References 10 publications

Update on brown tumor of hyperparathyroidism

Update on brown tumor of hyperparathyroidism

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

Brown Tumors: The Hidden Face of Primary and Renal Hyperparathyroidism Amid Real-Life Settings

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