Brown tumor of the cervical spine with primary hyperparathyroidism: A case report and literature review.

RATIONALE: Brown tumor (BT), an uncommon focal lytic bone tumor, is a non-neoplastic and reactive process caused by increased osteoclastic activity and fibroblastic proliferation in primary or secondary hyperparathyroidism. Vertebral tumor causing neural compression is relatively rare, especially in the cervical spine. PATIENT CONCERNS: A 29-year-old man developed neck pain and arm radicular pain 4 months ago, with the level of serum calcium significantly higher than normal. Computed tomography scan of the cervical spine revealed an expansile lytic lesion occupying the C6 body, left pedicle, and left lamina of C5-6. DIAGNOSES: Osteoclastoma according to imaging and histopathological results. INTERVENTIONS: A laminectomy of C5-6 was performed. OUTCOMES: One month later, he was re-hospitalized due to nausea and vomiting and the serum calcium, was still, kept at a high level. Additionally, the parathormone (PTH) was greatly higher than normal. BT with primary hyperparathyroidism due to the parathyroid tumor was considered. After the surgery of the right parathyroid gland was performed, serum calcium and PTH both decreased, and computed tomography showed good recovery. LESSONS: BTs might be misdiagnosed as other giant cell tumors, thus when giant cell tumors are considered, serum calcium and PTH examination may be needed to exclude BTs.

Misdiagnosis of brown tumour caused by primary hyperparathyroidism: a case report with literature review.

BACKGROUND: Brown tumour is a rare tumour-like lesion of the bone, which is considered as an end-stage lesion of abnormal bone metabolism caused by persistently high parathyroid hormone (PTH) levels. Brown tumour can be found in any part of the skeleton; in some cases, it can occur in multiple bones and can be easily misdiagnosed as a metastatic tumour. CASE PRESENTATION: We report the case of a 44-year-old man who presented to the Department of Oncology in our hospital with a 2-month history of local pain in his left shoulder joint. The initial diagnosis was an aneurysmal bone cyst by biopsy, for which the patient underwent tumour resection surgery. The diagnosis of a malignant tumour was made again following postoperative pathological examination. The pathological sections and all clinical data were sent to the Department of Pathology of the First Affiliated Hospital of Sun Yat-sen University; the diagnosis made there was brown tumour. His blood PTH level was 577 pg/ml (15-65 pg/ml). Colour Doppler ultrasonography of the parathyroid gland suggested a parathyroid adenoma. For further treatment, the left parathyroid adenoma was removed by axillary endoscopic resection. Postoperatively, a pathologic examination was performed, and the diagnosis of a parathyroid adenoma was confirmed. One year after the surgery, the left humerus was completely healed, and the left shoulder joint had a good range of movement. CONCLUSIONS: In summary, histopathological diagnosis is not sufficient for the diagnosis of brown tumours. A comprehensive analysis combining clinical symptoms with findings of imaging and laboratory tests is also required. Generally, the treatment of brown tumour includes only partial or complete resection of the parathyroid glands. However, when the tumour is large, especially when it involves the joint, surgery is indispensable.

Manifestations of hyperparathyroidism in the jaws: Concepts, mechanisms, and clinical aspects.

Hyperparathyroidism is one of the most common endocrine disorders worldwide. In countries where routine biochemical screening is not common, symptomatic hyperparathyroidism predominates. Its manifestations include skeletal alterations, calcification of soft tissues, kidney stones, and functional alterations in other systems. Notably, jaw alterations can be the first clinical sign of hyperparathyroidism, including brown tumor, renal osteodystrophy, osteitis fibrosa, and leontiasis ossea, and knowing such conditions is of core importance for the multidisciplinary diagnosis and management of hyperparathyroidism. We aimed to perform a concise review, systematizing the concepts and mechanisms underlying hyperparathyroidism and associated gnathic alterations. In addition, a detailed description of the clinical aspects of the jaw manifestations is presented.

Primary hyperparathyroidism misdiagnosed as metastatic disease, with trans-oral endoscopic surgical management.

Primary hyperparathyroidism is not so uncommon a disease. In high-income countries, with the introduction of routine biochemical testing in 1980s, it is diagnosed early, and the majority are seen as asymptomatic disease. However, in India, a severe symptomatic form is still seen, and many times patients are undiagnosed, thus continuing to suffer debilitating disease. Here, we report such a patient who was being treated as having bony metastases from an unknown primary source, when in fact he had osteitis fibrosa cystica due to primary hyperparathyroidism and was cured after surgery.

[The case of late diagnosis of giant parathyroid adenoma in combination with fibrocystic osteitis and brown tumor of the upper jaw: a case report].

In this case report the authors inform about late diagnosis of giant adenoma of the parathyroid gland with primary hyperparathyroidism (PHPT) and the development of fibrocystic osteitis with a «brown¼ tumor of the upper jaw. The patient has been under the care endocrinologist with type 2 diabetes mellitus and multinodular goiter for 8 years.The last 5 years there was a clinical manifestation of PHPT, but the diagnosis was made by an oncologist after the detection of a «brown¼ tumor of the upper jaw. According to multispiral computed tomography and scintigraphy with 99mTc-MIBI, a focal lesion was found in the upper jaw on the right, lytic foci in the bones of the cranial vault, pelvis, lower extremities, ribs on the right, as well as a giant parathyroid adenoma on the right. According to the increased risk of the patient having a malignant neoplasm of the parathyroid gland, an extended surgical treatment of PHPT in the enblock volume was carried out with the achievement of remission of the PHPT. This clinical case illustrates a variant of the severe course of PHPT with the development of such a rare complication as fibrocystic osteitis and demonstrates the importance of timely diagnosis.

Coexistence of brown tumors due to secondary hyperparathyroidism and prostate cancer in a patient.

We present the case of a 65-year-old man with brown tumors due to secondary hyperparathyroidism. Magnetic resonance imaging of the pelvis showed multiple lesions with expansive bone appearance. Additionally, prostate cancer was diagnosed during this time. For this reason, differential diagnosis was performed through biopsy of the right iliac bone lesion. Brown tumors are caused by osteoclastic activity and fibroblast proliferation; the differential diagnosis of these bone lesions includes giant tumors, metastases, Paget's disease, and paraneoplastic syndrome with high levels of parathyroid hormone-related peptide (PTHrP). This case report describes the coexistence of two pathologies that could explain these images. In this report, we present a case of a 65-year-old man with brown tumors due to secondary hyperparathyroidism and prostate cancer. In this setting, histologic confirmation is recommended.

Severe hypercalcemia and a pelvic brown tumor in an adolescent with primary hyperparathyroidism: a case report.

BACKGROUND: Primary hyperparathyroidism may present in a myriad of manners, varying from an incidental asymptomatic biochemical finding to gastrointestinal, psychiatric, renal, and bone manifestations. While hyperparathyroidism remains a rare diagnosis in the pediatric population, the initial approach to diagnosis and management of hypercalcemia in children is imperative for the general pediatrician. Herein, we describe an adolescent who presented with a lytic bone lesion and severe, symptomatic hypercalcemia due to primary hyperparathyroidism. CASE PRESENTATION: A 14-year-old male presented with vomiting, constipation, abdominal pain, and lethargy. He had an elevated total corrected calcium of 4.3 mmol/L. He was found to have a large pelvic lytic tumor consistent with a brown tumor due to primary hyperparathyroidism. He received pharmacologic therapy for stabilization of his hypercalcemia, including intravenous saline, intravenous bisphosphonates, and calcitonin. He subsequently received definitive therapy via parathyroidectomy and his post-operative course was complicated by hungry bone syndrome. Long-term follow-up has found full resolution of the lytic lesion and restored calcium homeostasis. CONCLUSIONS: We present this case to highlight the possible presentations of hypercalcemia and hyperparathyroidism that are essential for a general pediatrician to recognize to ensure prompt diagnosis and management. Evaluation for hypercalcemia should be considered in patients with suggestive symptoms and physical exam findings. To our knowledge, this patient represents the first reported pediatric case of a pelvic brown tumor in an adolescent. While the multi-systemic complications of hyperparathyroidism may be quite severe, swift and appropriate management may mitigate these clinical outcomes.

Primary hyperparathyroidism presenting as a brown tumor in the mandible: a case report.

BACKGROUND: Primary hyperparathyroidism is characterized by hypercalcemia and elevated or inappropriately normal serum levels of parathyroid hormone. Brown tumor of bone is a rare non-neoplastic lesion resulted from abnormal bone metabolism in hyperparathyroidism. However, nowadays, skeletal disease caused by primary hyperparathyroidism is uncommon. We report a case of brown tumor in the mandible as the initial exhibition of primary hyperparathyroidism associated with an atypical parathyroid adenoma. CASE PRESENTATION: The patient was a 49-year-old female, she had a pain mass on the right mandible a year ago and was treated with root canal therapy and marginal resection. After seven months, the mass recurred and enlarged. Enhanced CT scan, laboratory examination, Ultrasonography, 99mTc-MIBI SPECT-CT scintiscan and pathological examination were used to confirm the diagnosis of brown tumor. The patient's symptom improved after parathyroidectomy. CONCLUSIONS: 99mTc-MIBI SPECT/CT scintigraphy is a highly sensitive examination of the localization diagnosis of hyperparathyroidism. Brown tumors should be considered in the differential diagnosis of osteolytic lesions to avoid unnecessary and harmful interventions.

Osteítis fibrosa quística en paciente con hipercalcemia asintomática / Osteitis fibrosa cystica in a patient with asymptomatic hypercalcemia

La hipercalcemia es un hallazgo casual frecuente en el ámbito de la Atención Primaria, así como en los servicios de Urgencias. Valores poco elevados de calcemia o una instauración gradual de la misma suelen pasar clínicamente desapercibidos, por lo que su diagnóstico en pacientes asintomáticos es, con frecuencia, incidental. Nuestro objetivo principal en el diagnóstico diferencial de la hipercalcemia es determinar el mecanismo subyacente, sabiendo que el hiperparatiroidismo primario (HPP), junto con las neoplasias malignas, es responsable de hasta el 90 % de los casos; dichas entidades deben diferenciarse temprano para proporcionar al paciente un tratamiento óptimo y un pronóstico preciso. En nuestro trabajo presentamos el caso clínico de una mujer de 87 años, con antecedentes de hipercalcemia asintomática sostenida, que consulta por dolor e impotencia funcional no traumática del brazo izquierdo. En la prueba de imagen se observó una fractura del húmero proximal izquierdo sobre una estructura ósea alterada con grandes áreas quísticas "en sacabocados", entidad que recibe el nombre de osteítis fibrosa quística y que, aunque poco frecuente, es específica del HPP; en este caso, y tras completar el estudio, se vio que era debido a un adenoma paratiroideo único, responsable de hasta el 85 % de los casos del HPP. El único tratamiento definitivo, la paratiroidectomía, fue rechazado por la paciente, en quien se inició así tratamiento farmacológico, presentando hoy en día una buena evolución clínico-analítica. Las recomendaciones para los pacientes que no se someten a cirugía paratiroidea incluyen el control de las concentraciones séricas de calcio y densitometría ósea anuales

Hypercalcemia is a common chance finding in the primary care setting, as well as in emergency services. Low values of calcemia or its gradual establishment usually go clinically unnoticed, so the diagnosis is often incidental in asymptomatic patients. Our main objective in the differential diagnosis of hypercalcemia is to determine the underlying mechanism, knowing that primary hyperparathyroidism (PHPT) together with malignant neoplasms are responsible for up to 90% of the cases; these conditions must be differentiated early to provide the patient with optimal treatment and accurate prognosis. In our work, we present the clinical case of an 87-year-old woman with a history of sustained asymptomatic hypercalcemia, who presents with pain and non-traumatic functional deficit of the left arm. The imaging test showed a left proximal humerus fracture on an altered bone structure with large "punched-out" cystic areas, an entity that is called osteitis fibrosa cystica and that, although rare, is specific to PHPT; in this case, and after completing the study, it was seen that it was due to a parathyroid adenoma, responsible for up to 85% of cases of PHPT. The only definitive treatment, parathyroidectomy, was rejected by the patient. Therefore pharmacological treatment was initiated, presenting a good clinical-analytical evolution. Recommendations for patients who do not undergo parathyroid surgery include annual monitoring of serum calcium concentrations and bone densitometry

Solid aneurysmal bone cyst of the humerus mimics metastasis or brown tumor.

Solid aneurysmal bone cyst (ABC) is a rare subtype of ABC that most commonly involves the small bones of the hands or feet. We present a case of a solid ABC of the distal humerus in a 52-year-old man with a history of chronic kidney disease and renal cell carcinoma. On imaging with plain radiographs, CT, and MRI, this expansile lucent lesion with solid internal enhancement had an appearance that overlapped with metastasis or brown tumor of hyperparathyroidism. On 18F-FDG PET-CT, this lesion was hypermetabolic with an SUVmax of 9.9. Only 37 cases of solid ABC have previously been reported to involve the long bones in the literature, and only 4 in the humerus. We review the clinical, imaging, and histopathological findings and differential diagnosis of solid ABC, and highlight the usefulness of identifying the USP6 gene rearrangement on FISH to distinguish this lesion from other lesions with secondary ABC formation.

Osteolytic lesions (brown tumors) of primary hyperparathyroidism misdiagnosed as multifocal giant cell tumor of the distal ulna and radius: a case report.

BACKGROUND: Brown tumors represent a rare clinical manifestation reported in approximately 3% of patients with primary hyperparathyroidism and correspond to radiologically osteolytic lesions with well-defined borders in different parts of the skeleton. CASE PRESENTATION: We report the case of a 53-year-old white man who presented to our hospital with osteolytic lesions of his distal ulna and radius, causing pain and swelling of 2-month duration. A subsequent biopsy revealed histological features consistent with giant cell tumor and a complete resection of his distal ulna was followed, along with curettage and cementoplasty of the distal radial metaphysis. Two weeks later, he was re-admitted with diffuse musculoskeletal soreness, anorexia, constipation, nausea, and localized abdominal pain and multiple osteolytic lesions on plain radiographs. A histopathological examination of the ulna and radius specimens showed similar findings and, given the multifocality, brown tumors related to primary or secondary hyperparathyroidism was included in the differential diagnosis. A laboratory examination showed high total serum calcium (14.5 mg/dl) and low serum phosphorus and 25-hydroxyvitamin D levels. Primary hyperparathyroidism was suspected and confirmed by the elevated parathyroid hormone levels of 1453 pg/mL. At radiological work-up, using computed tomography, ultrasonography, and parathyroid subtraction technetium-99m sestamibi scintigraphy, a 4.5 × 2.5 × 3.2 cm mass emanating from the right lobe of his thyroid gland was detected, displaying extensive uptake in the right lower parathyroid gland. After appropriate medical support including hyperhydration and high doses of diuretics and diphosphonates, his laboratory profile normalized and he underwent total thyroidectomy with removal of the parathyroid glands. Our patient is now recovering 12 months after surgery, with normal values of serum parathyroid hormone and calcium levels. The lytic bone lesions have almost disappeared and no other additional orthopedic intervention was necessary. CONCLUSIONS: The present case report emphasizes the need of inclusion of brown tumors in the differential diagnosis of multifocal osteolytic bone lesions, in order to avoid harmful surgical interventions. Laboratory testing of serum phosphate, calcium levels, and parathyroid hormone levels should always be included in the routine survey of patients with multifocal osteolytic lesions.

Brown tumor of the jaws as a manifestation of tertiary hyperparathyroidism: A literature review and case report.

Brown tumor of the jaws is a manifestation of hyperparathyroidism consisting of osteolytic lesions that show proliferation of multinucleated giant cells in the maxilla and/or mandible. Differential diagnosis of these lesions from local central giant-cell granuloma is mandatory for the correct treatment of the patient. Radiographic and histopathological exams of the jaw lesion are not sufficient to determine the diagnosis, which requires laboratory tests including serum levels of calcium, alkaline phosphatase, parathyroid hormone (PTH) and phosphate, and radiographic examination of other bones as well, such as hand-wrist, pelvis, and femur. We present here a brief literature review focusing on the clinical and radiographic features, diagnostic criteria and treatment of brown tumor and also report a case of the disease affecting the jaw.

[Disseminated osteolytic lesions in a 28-year-old refugee]. / Disseminierte osteolytische Läsionen bei einem 28-jährigen Flüchtling.

A 28-year-old Syrian refugee presented with right-sided knee pain and progressive deterioration of the general condition over the past months. Laboratory diagnostics revealed severe hypercalcemia due to primary hyperparathyroidism, and computed tomography (CT) scanning demonstrated disseminated osteolytic lesions throughout the skeleton. Histologically, these lesions were characterized by multinuclear giant cells (defining these lesions as so-called brown tumors). Finally, surgical removal of a jugular mass allowed the histopathologic diagnosis of a sporadic parathyroid carcinoma. In the patient, this condition was associated with a mutation in the HPRT2 gene locus.

A patient with a history of breast cancer and multiple bone lesions: a case report.

BACKGROUND: Long-term severe hyperparathyroidism leads to thinning of cortical bone and cystic bone defects referred to as osteitis fibrosa cystica. Cysts filled with hemosiderin deposits may appear colored as "brown tumors." Osteitis fibrosa cystica and brown tumors are occasionally visualized as multiple, potentially corticalis-disrupting bone lesions mimicking metastases by bone scintigraphy or 18F-fluorodeoxyglucose positron emission tomography. CASE PRESENTATION: We report a case of a 72-year-old white woman who presented with malaise, weight loss, and hypercalcemia. She had a history of breast cancer 7 years before. The practitioner, suspecting bone metastases, initiated bone scintigraphy, which showed multiple bone lesions, and referred her to our hospital for further investigations. Laboratory investigations confirmed hypercalcemia but revealed a constellation of primary hyperparathyroidism and not hypercalcemia of malignancy; in the latter condition, a suppressed rather than an increased value of parathyroid hormone would have been expected. A parathyroid adenoma was found and surgically removed. The patient's postoperative course showed a hungry bone syndrome, and brown tumors were suspected. With the background of a previous breast cancer and lytic, partly corticalis-disrupting bone lesions, there was a great concern not to miss a concomitant malignant disease. Biopsies were not diagnostic for either malignancy or brown tumor. Six months after the patient's neck surgery, imaging showed healing of the bone lesions, and bone metastases could be excluded. CONCLUSIONS: This case shows essential differential diagnosis in a patient with hypercalcemia and multiple bone lesions. Whenever multiple, fluorodeoxyglucose-avid bone lesions are found, malignancy and metabolic bone disease should both be included in the differential diagnosis. Fluorodeoxyglucose-avid and corticalis-disrupting lytic lesions also occur in benign bone disease. There may be very few similar cases with heterogeneous and widespread bone lesions reported in the literature, but we think our patient's case is particularly remarkable for its detailed imaging and the well-documented course.

More than just tissue diagnosis in a patient with maxillofacial bony lesions and hypercalcemia.

Brown tumors are a definitive feature of hyperparathyroidism. They are well-demarcated osteolytic lesions commonly in the appendicular skeleton. Primary hyperparathyroidism is typically suggested by hypercalcemia and hypophosphatemia on routine labs. Much more rarely do these cases present with a craniofacial mass. Here we investigate a unique presentation of terminal stage primary hyperparathyroidism with a growing maxillary mass emphasizing the importance of a broad differential diagnosis and key diagnostic studies. Hyperparathyroidism can present in very unique ways. As otolaryngologists in the frontline, we must think beyond just tissue diagnoses so that appropriate and expedited care may be implemented. Laryngoscope, 127:1318-1321, 2017.