Brown Tumor Research Articles

Primary hyperparathyroidism revealed by multiple brown tumors: A case report

Searchable abstracts of presentations at key conferences in endocrinology ISSN 1470-3947 (print) | ISSN 1479-6848 (online)

Abstract #1406955: Two Parathyroidectomies in the Third Trimester for Severe Primary Hyperparathyroidism (PHPT) Resulting in Distinct Maternal and Fetal Outcomes

Parathyroidectomy (PTX) is well-tolerated in the second trimester of pregnancy in women with severe PHPT. We present two women requiring PTX in the third trimester of pregnancy due to the severity of the disease. Case 1: 30 y.o female presented at 33 weeks’ gestation with leg pain and severe PTH-mediated hypercalcemia; serum corrected calcium 4.2mmol/L (NR: 2.15-2.6), PTH 147.9 pmol/L (NR:1.6-6.9). She was symptomatic for 2 yrs prior and had a history of nephrolithiasis; the renal US showed 10-20 non-obstructing renal calculi. Femur X-ray demonstrated an aggressive lesion, identified as a brown tumor, on histopathology with additional bone lesions on skeletal survey. Severe hypercalcemia was managed with IV fluids, calcitonin and cinacalcet 30 mg BID. Neck-CT showed a 4cm parathyroid adenoma with no overt malignant features. Subtotal PTX was performed at 33w2d, and PTH dropped to 4.7pmol/L postop. Histopathology reported atypical parathyroid adenoma. Post PTX patient went into premature rupture of membranes, c-section done at 34 weeks’ gestation. Day 8 post PTX, the patient developed hungry bone syndrome, she also had a mechanical fall and sustained a pathological fracture of the femur, the site of brown tumor. Baby was admitted to NICU with hypoparathyroidism, resolved at 3 months of age. Case 2: 32 y.o female presented at 27 weeks’ gestation with incidental hypercalcemia and high PTH. Nausea and vomiting worsened from the 18th week of gestation. No end-organ involvement. Corrected calcium 3.04 mmol/L, PTH 17.1 pmol/L. Managed with IV fluids, and remained symptomatic with no improvement in hypercalcemia. PTX for parathyroid adenoma was performed at 30 weeks’ gestation with excellent maternal and fetal outcomes. PTH dropped to 2.0 pmol/L postop. Had spontaneous vaginal delivery at 39 weeks’ gestation, no neonatal hypocalcemia. Baby has normal milestones. Biochemical workup for Multiple Endocrine Neoplasia type1 (MEN1) was unremarkable for both patients, DNA testing is outstanding. The duration of PHPT, the severity of hypercalcemia, and the involvement of end-organ damage are critical factors that may influence both maternal and fetal outcomes in women with PHPT who undergo PTX in the third trimester.

Brown Tumor Related to Primary Hyperparathyroidism Mimicking Malignity in the Mandible: Case Report and Literature Review

Brown Tumor Related to Primary Hyperparathyroidism Mimicking Malignity in the Mandible: Case Report and Literature Review

Primary hyperparathyroidism in an adolescent presenting with genu valgus progressing to extensive bone disease; a case report

BackgroundPrimary hyperparathyroidism which is rare in adolescents presents commonly with non-specific symptoms and systemic complaints. Though there are few reported cases of genu valgus, genu valgus progressing to extensive bone disease despite mildly elevated calcium had not been reported before.Case presentationA 12-year-old male had been evaluated for bilateral (left > right) genu valgus and short stature. Serum calcium and phosphate levels had been normal. X-ray of the femora and pelvic bones had not shown additional abnormalities. Valgus deformity progressed despite left femoral plating, and a left distal femoral medial closed wedge osteotomy had been performed at 15 years. Plain imaging at that time had shown localised osteopaenia. At the age of 17 years, he developed multiple fragility fractures of his left hip rendering him wheelchair-bound. Further evaluation revealed a serum PTH level of 2571 (10–65) pg/mL with calcium of 2.82 (2.2–2.6) mmol/L and inorganic phosphate of 1.7 (2.2–4.7) mg/dL. The lumbar spine DXA scan showed a Z-score of -5.8. A left parathyroid adenoma was localised and there was evidence of hyperparathyroid bone disease including brown tumours. He underwent left parathyroidectomy and left thyroid lobectomy after which his PTH level dropped to 4.03 pg/mL. He developed hypocalcaemia which was managed successfully with calcium and alfacalcidol replacement.ConclusionsPrimary hyperparathyroidism can present with genu valgus in adolescents. Initial normocalcaemia which could be due to concomitant vitamin D deficiency could mask this leading to delayed diagnosis until severe irreversible bone disease ensues.

The pathology of brown tumor in a-36-year-old man on maintenance hemodialysis

The pathology of brown tumor in a-36-year-old man on maintenance hemodialysis

Clinical Images: Increased bone metabolism in secondary hyperparathyroidism.

The patient, a 47-year-old man, was diagnosed as having severe hyperparathyroidism secondary to end-stage kidney disease (ESKD), the underlying cause being leukocytoclastic vasculitis with IgA nephritis and minimal-change glomerulonephritis, which was confirmed by kidney biopsy in 2008. The patient had been receiving hemodialysis since 2014 and had a history of multiple fractures. His treatment included azathioprine, low-dose prednisolone, calcium carbonate, vitamin D, sevelamer, cinacalcet, torasemide, carvedilol, amlodipine, moxonidine, simvastatin, low molecular weight heparin, and erythropoietin. After a sudden transitory episode of vision loss with increased C-reactive protein (18.1 mg/liter) and response to glucocorticoids, 18F–positron emission tomography/computed tomography (18F-PET/CT) evaluation was performed to rule out systemic vasculitis. Ophthalmologic examination was negative for arteritic ischemic optic neuropathy, ophthalmoparesis, loss of visual field, and diplopia. Imaging with 18F-PET/CT revealed enhanced fluorodeoxyglucose uptake in the skeleton (A and B) and thickening of cortical bone such as in the femurs (C). Bone loss as osteolytic lesions in the calvaria, increased uptake in the setting of diffuse skull thickening with loss of distinction between the inner and outer table (D), and increased uptake in mandible bones (E) could be seen. There was no evidence for vasculitis. ESKD is associated with a complex remodeling of bone, summarized as renal osteopathy (1). This might include bone resorption, periosteal reaction, brown tumors, osteoporosis, osteosclerosis, osteomalacia, and soft tissue/vascular calcifications. Imaging may reveal any number of these conditions (2, 3). In our patient, 18F-PET/CT revealed osteolytic lesions, which were considered brown tumors and interpreted as osteitis fibrosa cystica. The patient was hypocalcemic (adjusted serum calcium 1.90 mmoles/liter), with elevated alkaline phosphatase (758 IU/liter) and massively elevated parathyroid hormone levels (1,872 pmoles/liter), indicating severe secondary hyperparathyroidism. These 18F-PET/CT images reflect the widespread, strongly induced metabolic activity of bone in secondary hyperparathyroidism during ESKD. Disclosure Form Please note: The publisher is not responsible for the content or functionality of any supporting information supplied by the authors. Any queries (other than missing content) should be directed to the corresponding author for the article.

Renal osteodystrophy and brown tumours of the jaw associated with tertiary hyperparathyroidism in the context of cystinosis: a case report

Chronic kidney disease is one of the main manifestations of cystinosis that may lead to hyperparathyroidism. Although head and neck involvement in hyperparathyroidism is rare, lesions can occur in the jaws and cause serious functional handicap. We presented the case of a 24-year-old woman with congenital cystinosis and chronic renal failure complaining from symptomatic expansion of the jawbones. Medical imaging showed radiolucent lesions particularly in anterior hard palate and body of mandible. A diagnosis of renal osteodystrophy and brown tumours related to tertiary hyperparathyroidism was established. Following a subtotal parathyroidectomy and stabilization of parathyroid hormone levels, a conservative mandibular osteoplasty was undertaken to increase the available space for tongue movements. Although non-surgical management remains the primary modality for the treatment of brown tumours, adjunctive surgical approach may be considered in cases of severe functional impairment. It is imperative that underlying systemic imbalances be addressed prior to any maxillofacial intervention to prevent relapse.

Diffuse Calvarial Hyperstosis

Calvarial hyperstosis can be an idiopathic benign finding or secondary to a metabolic pathology. We herein describe a case of diffuse calvarial hyperstosis. A 26-year-old man known to have end-stage renal disease on regular hemodialysis, tertiary hyperparathyroidism, extensive brown tumors, and severe developmental impairment with skeletal deformities was referred to us for macrocephaly. On examination, the patient was chairbound, with speech and motor developmental delay, and frontal bossing. Brain computed tomography revealed diffuse hyperstosis of the calvarium and facial bones expansion with multiple sclerotic and lytic areas, causing subsequent narrowing of the basilar skull foramina. Brain magnetic resonance imaging demonstrated an extensive expansile bone marrow abnormality in the calvarium and skull base. There was mild generalized prominence of cortical sulci and ventricular system. The findings were in keeping with his known hypermetabolic state and tertiary hyperparathyroidism. The patient was managed conservatively with regular follow-up in the clinic.

Brown tumors in nuclear medicine: a systematic review

Brown tumors (BT) are abnormal bone-repair processes and a consequence of hyperparathyroidism. The diagnosis of these lytic lesions in nuclear medicine, while a challenge, is not so rare, because functional imaging is used both in the management of cancer and hyperparathyroidism. The main objective of this review is to summarize the knowledge and the evidence concerning BT and the different imaging modalities in nuclear medicine. A systematic review was performed in Embase, PubMed and Google Scholar from 2005 to 2022. We included articles describing BT in the following imaging modalities: [18F]-fluorodeoxyglucose PET/CT, [18F]-fluorocholine or [11C]-fluorocholine PET/CT, [99mTc]-Sestamibi scintigraphy, bone scan, [18F]-sodium fluoride PET/CT, [68Ga]-FAPI PET/CT; [68Ga]-DOTATATE PET/CT; [11C]-methionine PET/CT. For each modality, appearance, avidity for radiotracer, available quantitative parameters and imaging evolution after parathyroidectomy were collected and analyzed. Fifty-two articles were included for a total of 392 BT lesions. If the diagnosis of BT is evoked on a known lesion, performing a [18F]-fluorocholine PET/CT imaging seems the most appropriate. In [18F]-fluorodeoxyglucose, [18F]-fluorocholine, [18F]-sodium fluoride PET/CT and bone scan, BT can mimic metastatic disease. BT uptakes appear reversible after parathyroidectomy, with a more or less rapid decrease depending on the imaging modality used.

Conservative Treatment in Impending Fracture of Solitary Brown Tumor of Tibia: A Case Report and Review of Literature

Background: Brown tumors, also known as osteitis fibrosa cystica, are focal benign bone lesions caused by an increased osteoclastic activity because of high levels of parathyroid hormones. The increase of parathyroid hormones can be primary hyperparathyroidism (HPT) and secondary HPT resulting from renal failure and vitamin D deficiency. Case presentation: A 26-year-old Thai female visited the clinic because of right leg pain and swelling for three weeks. Plain radiography revealed a solitary osteolytic lesion with an impending fracture at the anterior cortex of the right proximal tibia. The patient underwent a core needle biopsy. The initial histopathology report depicted a giant cell lesion. In addition, the patient had elevated serum parathyroid hormones. A parathyroid adenoma was discovered on a further investigation with contrast-enhanced computed tomography of the neck. The immunochemistry and molecular study report were consistent with a brown tumor. The patient underwent parathyroidectomy for her adenoma and was on weight-bearing restrictions for her leg. After three months of follow-up, she could bear weight on her right leg without pain, and radiographic evaluation showed restoration of the tibial cortex. Conclusion: Conservative management has a role in managing brown tumors of the lower extremities with impending pathologic fracture. Rapid diagnosis and removing the primary cause of parathyroid adenoma as soon as possible is a crucial part of the treatment.

Brown tumor of the rib associated with primary hyperparathyroidism misdiagnosed as metastatic deposit - A case report

Brown tumor is a rare skeletal manifestation where multiple giant cell rich bony lesions, arise as a direct result of either primary or secondary hyperparathyroidism. It is caused by excess osteoclastic activity and hemosiderin deposition because of uncontrolled parathormone secretion. Brown tumors usually involve mandible, maxilla, clavicle, ribs, and pelvic bones. Our case report describes the case of a 42-year-old lady who consulted our hospital for multiple bony lesions in ribs suspicious of metastasis and summarizes our experience in diagnosis of similar cases that we might come across in the future.

Primary hyperparathyroidism: Brown tumor in hand, foot, and femur

Abstract Introduction Bone fractures due to primary hyperparathyroidism/brown tumor can still be seen today, albeit rarely. Clinical Case A 47-year-old migrant woman had a right femur fracture after a simple fall. The patient's laboratory values were found as calcium 16.3 (8.4–10.2) mg/dl, phosphorus 2.5 (3.2–5.5) mg/dl, Alkaline phosphatase: 1131 (40–150) U/L, Parathormone 1711 (15–65) pg/ml. A 40×30 mm parathyroid adenoma was detected in the lower left side of the patient, who was suspected of having primary hyperparathyroidism. Parathyroid adenoma in the same localization was also observed in neck tomography. At the same time, a brown tumor was detected on the hand and foot bones. The patient underwent a left parathyroid adenomectomy. Intravenous calcium infusion was given to the patient who developed hungry bone syndrome in the postoperative period. Calcium, phosphorus, and parathormone levels of the patient who was treated for hungry bone syndrome returned to normal. Later on, the patient underwent a right femoral prosthesis operation. Conclusion Brown tumor is often seen in the bones of the hands, feet, and long bones.

Primary hyperparathyroidism and skull bone: Dicle sign

Abstract Introduction In primary hyperparathyroidism, a brown tumor may be seen due to excessive osteoclastic activity. We will share the unusual localization of this tumor-like formation that primarily affects the lower and upper extremities. Clinical Case A 20-year-old female patient presented with complaints of weakness, headache, bone pain, and difficulty walking. He had a history of surgery for a pathological femur fracture in the past. In the evaluation, a diagnosis of primary hyperparathyroidism due to ectopic parathyroid adenoma was made. Brown tumor-like formations were also observed in the skull bone in the cranial tomography taken due to severe headache. An adenomectomy was performed on the patient whose parathyroid adenoma localization was detected in the scintigraphic examination. The patient, who developed post-operative hungry bone syndrome, was discharged approximately one month later due to the need for intravenous calcium. It lasted about one year after the patient's oral calcitriol and calcium treatment. She was followed up without treatment. At her last follow-up in June 2022, calcium, phosphorus, and parathormone levels were normal. The patient had no complaints. Conclusion Brown tumor is often seen in the bones of the hands, feet, and long bones. We detected it for the first time in the skull bone.

Brown Tumors of the Jaws: A Retrospective Study.

(1) Describe the clinical spectrum, imaging features, management, and outcome of brown tumors (BT) of the jaws. (2) Determine via a literature review the facts and controversies around the characteristics and management of this lesion. Our study was approved by the institutional committee of Sahloul University Hospital in accordance with the ethical standards of the Declaration of Helsinki. Sixteen patients with BT in the context of a primary, secondary, and tertiary hyperparathyroidism were treated and followed up during their recovery. This study reports 16 patients with a mean age of 48 years old. Brown tumor lesions were associated with primary hyperparathyroidism in 6 cases (38%), secondary hyperparathyroidism to chronic kidney failure in 5 cases (31%), and tertiary hyperparathyroidism in the context of a long lasting CRI in 5 cases. Their location was maxillary in 7 cases, mandibular in 5 cases, and bimaxillary in 4 cases. The treatment consisted of parathyroidectomy in 13 patients, maxillary resection in 3 cases, and vitamin D treatment in 2 cases. Favorable outcomes, characterized by tumor regression, were reported in 9 cases, whereas unfavorable evolution was observed among 7 patients. Parathyroidectomy is the gold standard treatment for primary hyperparathyroidism. It resulted in a total regression in all our cases. Regarding secondary hyperparathyroidism, blood screening and chronic renal insufficiency follow-up are critically valuable to detect this condition at an early stage, hence increasing the success rate of brown tumor regression. Our secondary and tertiary hyperparathyroidism results were miscellaneous; although it is important to emphasize the importance of the chronic renal insufficiency management to ensure a success. Brown tumors should be included in the differential diagnosis of giant cell lesions. Parathyroidectomy is usually sufficient to induce the total regression of the lesion in primary hyperparathyroidism cases. A more delicate approach is needed regarding secondary hyperparathyroidism. Meticulous control of chronic renal insufficiency is mandatory in addition to parathyroidectomy.

Massive brown tumors of the jaw: A case report

Brown tumors are non-neoplastic bone lesions caused by an abnormal remodeling of the bone that may occur with primary or secondary hyperparathyroidism. Their radiological aspect: lytic and aggressive can easily be misdiagnosed for a malignant origin hence the importance of knowing that diagnosis is to be considered through both clinical context and radiological semiology, which will be detailed via this case of a 32-year-old female patient with an end-stage kidney disease, admitted for facial disfiguration and palpable masses corresponding to brown tumors affecting the maxilla and the mandibular bone.

Leontiasis Ossea about a Case Treated at Chu Owendo

Introduction: Leontiasis ossea is the most severe bone complication of the maxillae of chronic renal failure. The objective of this work is to determine the clinical, paraclinical and therapeutic aspects of this condition. Observation: A 42-year-old patient with chronic anuric terminal renal failure dialysis for 12 years had consulted for multiple maxillo-mandibular swelling evolving for 2 years deforming the face with discomfort to chewing. The biological assessment was: PTH: 5252 pg/mL, Ca2+: 2.86 mmol/L, free T3: 4.16 pmol/L, free T4: 6.55 pmol/L, TSH: 3.13 U/mL, serum protein: 61 g/L, Creatinine: 835 μmol/L. The maxillofacial CT noted heterogeneous diffuse thickening of the cranial vault and bones of the facial massif evoking leontiasis ossea with right mandibular brown tumor. The biopsy diagnosed fibrous gingival epulis. The patient had undergone modeling surgery, under general anesthesia after dialysis, of the right mandibular tumor whose histological result was Leontiasis ossea. Conclusion: Treatment is modeling surgery under hemostatic control.

Brown tumor of the mandible - a possible clinical manifestation of primary hyperparathyreoidism

Introduction. One of the possible manifestations of primary hyperparathyroidism (PHPT) is the appearance of a benign bone tumor. We hereby present a case of a young woman whose first clinical manifestation of PHPT was a brown tumor of the mandible. Case report. A 27-year-old female patient was hospitalized at the Clinic for Endocrinology, Diabetes, and Metabolic Diseases due to problems occurring in the form of nausea, exhaustion, the feeling of suffocation, dysphagia, pain in the right ear and the right half of the cheeks, with suspected PHPT. Initial laboratory findings pointed out the high levels of parathyroid (PT) hormone (PTH) and calcium (Ca2+) ions, low levels of vitamin D, and increased parameters of bone metabolism with signs of osteopenia. Cone beam computed tomography revealed the presence of bilateral radiolucent lesions of the mandible. Scintigraphy verified a retrosternal hot focus consistent with PT adenoma. After parathyroidectomy was performed, there was a normalization of PTH values, bone metabolism parameters, and the Ca2+ values. Four months after parathyroidectomy, a significant regression of the mandibular tumefaction was confirmed, clinically and radiologically. Conclusion. Brown tumors are rare first clinical manifestations of PHPT. Owing to their histological similarities with other giant-cell lesions (GCLs), definitive diagnosis is sometimes difficult and is based on a correlation of pathohistological, radiological, and laboratory findings. Due to the spontaneous regression of bone lesions after treatment of the basic cause of PHPT, brown tumors should be considered in the differential diagnosis of any GCLs in order to avoid unnecessary surgical procedures.

Радионуклидная диагностика в определении сопутствующей патологии у онкологических больных

The aim of this study is to demonstrate the significance and necessity of radionuclide diagnostics of oncologic pathology of different nature on the instance of a clinical case of detection of multiple foci of secondary nature (mts) of parathyroid gland tumor by skeletal bone scintigraphy in the course of routine examination of a patient for breast cancer. The data from the medical records, protocols and radiation methods of examination of the patient were used as the study material. During a routine examination for metastases of breast cancer through radionuclide examination of the parathyroid glands and scintigraphic examination of the skeletal bones, pathologic foci of radiopharmaceutical (RP) hyperfixation were detected in the patient. On further computed tomography (CT) of the knee joints it was found that the picture may correspond to multiple secondary lesions of the skeletal bones on the background of parathyroid gland disease. After a week the patient underwent a biphasic radionuclide study of the parathyroid glands, which made it possible to assess the presence of obvious scintigraphic signs of hyperfunctioning adenoma of the left lower parathyroid gland. Further, a thyroid gland (TG) tissue biopsy study was performed, which showed that the tissue fragment on the section was light brown with the presence of a cyst filled with brown fluid, which may correspond to a brown tumor. Thus, a patient with main breast disease is likely to be diagnosed with other unrelated cancers. The key diagnostic method in this case will be a scintigraphic study with single photon emission computed tomography (SPECT), which provides the most accurate visualization of pathology, its description and prediction of further therapy both in outpatient and inpatient settings. For the majority of cancer patients it is advisable to conduct regular examination with the help of scintigraphy for the purpose of prevention and early detection of concomitant pathology.

Mandibular brown tumor revealing an ectopic parathyroid adenoma

The case report presents a 47-year-old female patient. She was suffering a painful clinical mandibular lesion. The histological diagnosis returned in favor of a brown tumor (osteitis fibrosa cystica). The patient did not present any signs of hypercalcemia. The biology report shows an hypercalcemia and an elevated parathyroid hormone (PTH) levels. The Sestamibi (methoxyisobutylisonitrile) scintigraphy returned in favor of an ectopic pathological parathyroid tissue in the upper mediastinal zone. The excision of this tissue revealed a parathyroid adenoma, which normalized the metabolic state and regressed the mandibular lesion. This case study highlights the importance of the dosage of parathyroid hormones and the calcemia in identifying the origin of osteitis fibrosa cystica.

Oral kavitede kitle ve eşlik eden insidental papiller tiroid karsinomu ile başvuran normokalsemik primer hiperparatiroidizm olgusu

Hyperparathyroidism (HPT) is an endocrine disorder characterized by high secretion of parathyroid hormone. Brown tumor is one of the skeletal manifestations of HPT. Its overall prevalence is low (about 0.1%). Involvement of facial bones is extremely rare. The mandible is involved in 4.5% of cases. Primary HPT (PHPT)with vitamin D deficiency worsens the clinical course. Parathyroid adenoma is detected in ectopic places with a rate of 8.5%. 0.2% is intrathyroidal, 2% is located in different neck regions, 4.1% is in the upper mediastinum, and 2.2% is in the lower mediastinum. The prevalence of papillary thyroid cancer (PTC) in PHPT patients has been reported to range from 2% to 15%. In the literature, incidentally detected papillary thyroid cancer in patients with hyperparathyroidism was detected in patients who underwent total thyroidectomy associated with intrathyroidal parathyroid adenoma.
 In this article, we present a case of incidental papillary thyroid carcinoma with ectopic parathyroid adenoma accompanied by severe vitamin D, and diagnosed during accompanying surgery following a brown tumor was considered after primary hyperparathyroidism was detected. However, the patient was admitted with a mass in the oral cavity, diagnosed before as a pyogenic granuloma with a delayed diagnosis without considering brown tumor because it was normocalcemic. It is the first case in the literature of papillary thyroid carcinoma detected incidentally with ectopic parathyroid adenoma in the thyrothymic region.