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.

Mandibular Brown Tumor as a Result of Secondary Hyperparathyroidism: A Case Report with 5 Years Follow-Up and Review of the Literature.

BACKGROUND: Brown tumor is a rare skeletal manifestation of secondary hyperparathyroidism. Although diagnosis of the disease is increasingly seen in early stages due to improved screening techniques, some patients still present in a progressed disease stage. The treatment depends on tumor mass and varies from a conservative approach with supportive parathyroidectomy to extensive surgical resection with subsequent reconstruction. CASE PRESENTATION: We report a case of extensive mandibular brown tumor in a patient with a history of systemic lupus erythematosus, chronic kidney disease, and secondary hyperparathyroidism. Following radical resection of the affected bone, reconstruction could be successfully performed using a free flap. CONCLUSIONS: There were no signs of recurrence during five years of close follow-up. Increased awareness and multidisciplinary follow-ups could allow early diagnosis and prevent the need for radical therapeutical approaches.

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.

Spinal Cοrd Compression Secondary to Brοwn Tumοr as First Manifestation of Primary Hyperparathyroidism.

BACKGROUND: Brown tumors (BTs) represent the typical nonmalignant lesions of hyperparathyroidism. Mandibles, ribs, and large bones are the most usual localization of BT. The diagnosis of these tumors requires biological and radiologic assessments. Their treatment is essentially based on parathyroidectomy. CASE DESCRIPTION: The present case report describes a patient with primary hyperparathyroidism who developed a cervical BT revealed by slow spinal compression. CONCLUSIONS: The brown tumor, when localized at the level of the spine, can be life-threatening and must be managed as soon as possible.

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.

Manejo quirúrgico de tumor pardo en metacarpo. Reporte de caso / Surgical management of Metacarpal brown tumor. A case report

El tumor pardo, también conocido como osteoclastoma ó como osteítis fibrosa quística, es un tumor lítico, que se presenta en hiperparatiroidismo (primario, secundario y terciario), aunque su presentación habitual es altamente invasiva, no tiene potencial de malignidad. Los tumores pardos en la mano son muy poco frecuentes y existen solo algunos reportes de casos. Presentamos un paciente masculino de 18 años con una tumoración dura, no móvil, adherida a planos profundos en región dorsal de la mano derecha sobre el cuarto metacarpiano, que además limita la flexión y extensión del cuarto dedo sin alterar su función neurovascular. El paciente fue sometido a resección de la tumoración que involucraba por completo al cuarto metacarpiano derecho, además se realizó un abordaje lateral directo en miembro pelvico izquierdo para tomar un injerto autólogo de peroné no vascularizado. Es importante la detección temprana de este tipo de tumores y se debe dar un adecuado seguimiento, ya que, al progresar, generan una destrucción ósea importante y el tratamiento se vuelve de mayor complejidad. En etapas tempranas, el manejo agresivo con resección y aporte óseo puede evitar secuelas funcionales. El uso de injerto no vascularizado de peroné de seis centímetros para la sustitución del cuarto metacarpiano por osteolísis secundaria a un tumor pardo es una alternativa adecuada de tratamiento que permite la preservación estético funcional de la mano.

The brown tumour, also known as osteoclastoma, or as osteitis fibrosa cystica, is a lytic tumour, which occurs in hyperparathyroidism (primary, secondary, and tertiary), although its usual presentation is highly invasive, has no potential for malignancy. Brown tumours of the hand are sporadic, and there are only few case reports. The case is presented of an 18-year-old male patient with a solid, non-mobile tumour, adhered to deep planes, in the dorsal region of the right hand over the fourth metacarpal. This also limited the flexion and extension of the fourth finger, but did not show alterations in the neurovascular function of the finger. The patient underwent a tumour resection that completely involved the right fourth metacarpal. A direct lateral approach was made in the left pelvic limb to perform a non-vascularised autologous fibular graft. Early detection of this type of tumour is important, and an adequate follow-up must be carried out, since when they progress, they generate significant bone destruction and the treatment becomes more complex. In early stages, aggressive management of resection and bone support can prevent functional sequelae.

A hemodialysis patient with bone disease after pregnancy: a case report.

BACKGROUND: Pregnancy is rare in women on hemodialysis. Recommendations for the treatment of secondary hyperparathyroidism (sHPT) and preservation of bone health in pregnant dialysis patients are lacking. CASE PRESENTATION: We present the case of a young woman with end-stage kidney disease (ESKD) due to lupus nephritis, who developed multiple brown tumors while on hemodialysis during her second pregnancy. During her first pregnancy sHPT was well controlled and no skeletal complications occurred. Before the second pregnancy she developed severe sHPT. During pregnancy, dialysis time was increased to 24 h per week, the patient was given oral calcitriol, and the dialysate calcium concentration was set at 1.5 mmol/l. In week 20 the patient complained about bone pain in her left hip. Magnetic resonance imaging revealed a cystic lesion compatible with a brown tumor. The baby was delivered in the 36th week by cesarean section. Further assessment identified multiple brown tumors of her skeleton, including the acetabulum, tibia, ribs, skull, thoracic spine and thumb. She required multiple orthopedic surgeries. Three months after pregnancy, etelcalcetide was started, which brought about a gradual improvement in her sHPT. CONCLUSIONS: This case demonstrates that the combination of pregnancy and severe sHPT in dialysis patients can have deleterious consequences for bone health.

Brown tumor of the thoracic spine presenting with paraplegia in a patient with peritoneal dialysis.

Secondary and tertiary hyperparathyroidism is an important problem of chronic kidney disease. Brown tumor is a benign, unusual, reactive lesion as a result of disturbed bone remodeling, from long-standing increase in parathyroid hormone level. Brown tumors may cause morbidity due to pressure symptoms on neural structures and spontaneous bone fractures. Herein, we presented a peritoneal dialysis patient with tertiary hyperparathyroidism under calcand calcitriol treatment for 4 years due to refusing of the parathyroidectomy operation. She admitted to hospital for sudden onset back pain with difficulty in gait and walking, and imaging studies showed an expansile mass lesion in the thoracic spine. She was operated for mass and diagnosed with brown tumor. After operation, she lost the ability of walking than become paraplegic and she underwent rehabilitation program. Preventive measures including calcitriol and cinacalcet may cause a modest decrease in parathyroid hormone levels but it should be remembered for the development of bone complications such as brown tumor formation in patients with moderate elevated PTH levels, especially those with tertiary hyperparathyroidism. Parathyroidectomy should be performed without delay in these cases.

The Use of Virtual Surgical-Guided Osteoplasty for Maxillofacial Brown Tumors.

A 28-year-old African American female with end-stage renal disease on dialysis secondary to preeclampsia presented to the office as a referral for large multifocal tumors of maxilla and mandible. Surgical pathology and laboratory findings were supportive of secondary hyperparathyroidism leading to multifocal brown tumors. She underwent osteoplasty after using virtual surgical planning to create stereolithic models to visualize the tumor and fabricate cutting guides to minimize the risk of injury to adjacent nerves and teeth. Brown tumors can be resistant to medical management with unreliable regression in size. With the advent of customized surgical guides, more precise and judicious surgery can be performed on these types of tumors safely.

Complex spinal fixation of a cervical vertebra Brown tumour: report of an unusual case.

Brown tumours affecting the cervical spine are a rare but recognised complication of renal failure-related secondary hyperparathyroidism. We present a case of a 26 year-old female with radiculopathy who was managed successfully with 360° cervical spine fixation and parathyroidectomy.

Suspected Malignant Lesion of the Hip Resolves after Parathyroidectomy: Osteitis Fibrosa Cystica (or Brown Tumor): A Case Report.

We present a case of osteitis fibrosa cystica (OFC), also known as "brown tumor." A 55-year old female was admitted to the hospital with several months of right hip pain that was becoming more severe. A malignant lesion of the pelvis was suspected after initial imaging studies. Significant hypercalcemia led to a diagnosis of hyperparathyroidism and ultimately a benign parathyroid adenoma. Surgical excision of the adenoma resulted in full resolution of the tumor in her pelvis as well as her pain. It is important to keep OFC, or brown tumor, in your differential diagnoses when presented with a bone lesion.

Expansive renal osteitis fibrosa: a case report.

Hyperparathyroidism (HPT) is an endocrine metabolic disorder characterized by increased secretion of parathyroid hormone. Untreated secondary HPT leads to renal osteodystrophy (ROD). Facial skeletal abnormalities in patients with ROD are rare. The purpose of this paper is to report a conservative surgical approach of exuberant osteitis fibrosa lesions in patient with chronic kidney disease. A 24-year-old female was referred to maxillofacial surgery department with giants ROD affecting palate, maxilla, and mandible, resulting in esthetic and functional impairment. The pathogeneses and multidisciplinary management of ROD are discussed with a brief literature review. Eight years after the conservative treatment of exuberant jaw lesions, no noticeable bone changes were observed in the patient. A multidisciplinary therapy is essential for correct diagnosis of ROD and optimal multimodality treatment. The conservative management was an efficient alternative for the success of the case reported.

Multiple brown tumors - a rare presentation in the modern era
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Chronic kidney disease (CKD) commonly evolves with disturbances in mineral and bone metabolism, currently defined as CKD-MBD. Management strategies have progressed over the years, but our knowledge regarding evaluation and treatment is still sparse. Herein, we describe a rare case of a hemodialysis patient with apparently fairly controlled hyperparathyroidism (HPTH), who developed multiple symptomatic brown tumors involving the scull, mandible, vertebrae, pelvis, and metacarpus. Parathyroidectomy allowed complete resolution of the bone lesions preventing disastrous consequences.
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Giant mediastinal parathyroid adenoma presenting as bilateral brown tumour of mandible: a rare presentation of primary hyperparathyroidism.

Hyperparathyroidism (HPT) is becoming increasingly common endocrinopathy in clinical practice. Nowadays, it is mostly diagnosed in subclinical or early clinical stage. Bony involvement in HPT has seen significant fall in incidence. Brown tumour of bone is exceptionally rare as a first manifestation of primary HPT (PHPT). Its radiological and histopathological features may be mistaken for other bony pathologies. If possibility of underlying HPT is overlooked the disease is bound to recur after surgery adding to morbidity of the patient. Here we present a case of bilateral brown tumour of mandible which was mistakenly treated as giant cell granuloma by surgical curettage. That the patient was harbouring an ectopic parathyroid adenoma with hypercalcemia causing non-specific symptoms was missed by the referring physician. This led to recurrence of the lesion. On subsequent evaluation, a giant mediastinal parathyroid adenoma causing PHPT was detected at our centre and was removed via mini sternotomy approach.

Brown tumor of secondary hyperparathyroidism: surgical approach and clinical outcome.

BACKGROUND: Secondary hyperparathyroidism is a frequent complication of chronic renal failure. The brown tumor is an unusual presentation of fibrous osteitis that represents a serious complication of renal osteodystrophy, affecting predominantly the hands, feet, skull, and facial bones. CASE REPORT: The aim of this paper is to describe the case of a 53-year-old female patient, with renal failure who has been on dialysis for 6 years and developed severe secondary hyperparathyroidism and brown tumor of the maxilla and mandible, confirmed by incisional biopsy. Parathyroidectomy was indicated as a result of rapid growth of the tumor and the maintenance of laboratory findings. Despite the normalization of serum parathyroid hormone and alkaline phosphatase, tumor regression was slow and patient's important functional and esthetic deficits persisted. Excision of the mandible tumor was conservative. Osteoplasty was recommended because during a 5-year follow-up there was regression of the lesion, decreased pain, bleeding, and tooth mobility.

Imaging findings of spinal brown tumors: a rare but important cause of pathologic fracture and spinal cord compression.

Brown tumors rarely develop in the spine, and neurological compromise is exceedingly uncommon. There is a growing body of literature describing brown tumors that involve the spine, but few emphasize the radiographic findings. In the present case, we illustrate the development and progression of biopsy-proven brown tumors leading to neurological compromise through radiographs, computed tomography, magnetic resonance, and nuclear imaging acquired over a 4-year span.

Bone disease in primary hyperparathyroidism.

Bone disease in severe primary hyperparathyroidism (PHPT) is described classically as osteitis fibrosa cystica (OFC). Bone pain, skeletal deformities and pathological fractures are features of OFC. Bone mineral density is usually extremely low in OFC, but it is reversible after surgical cure. The signs and symptoms of severe bone disease include bone pain, pathologic fractures, proximal muscle weakness with hyperreflexia. Bone involvement is typically characterized as salt-and-pepper appearance in the skull, bone erosions and bone resorption of the phalanges, brown tumors and cysts. In the radiography, diffuse demineralization is observed, along with pathological fractures, particularly in the long bones of the extremities. In severe, symptomatic PHPT, marked elevation of the serum calcium and PTH concentrations are seen and renal involvement is manifested by nephrolithiasis and nephrocalcinosis. A new technology, recently approved for clinical use in the United States and Europe, is likely to become more widely available because it is an adaptation of the lumbar spine DXA image. Trabecular bone score (TBS) is a gray-level textural analysis that provides an indirect index of trabecular microarchitecture. Newer technologies, such as high-resolution peripheral quantitative computed tomography (HR-pQCT), have provided further understanding of the microstructural skeletal features in PHPT.

Bone disease in primary hyperparathyroidism / Doença óssea no hiperparatiroidismo primário

Bone disease in severe primary hyperparathyroidism (PHPT) is described classically as osteitis fibrosa cystica (OFC). Bone pain, skeletal deformities and pathological fractures are features of OFC. Bone mineral density is usually extremely low in OFC, but it is reversible after surgical cure. The signs and symptoms of severe bone disease include bone pain, pathologic fractures, proximal muscle weakness with hyperreflexia. Bone involvement is typically characterized as salt-and-pepper appearance in the skull, bone erosions and bone resorption of the phalanges, brown tumors and cysts. In the radiography, diffuse demineralization is observed, along with pathological fractures, particularly in the long bones of the extremities. In severe, symptomatic PHPT, marked elevation of the serum calcium and PTH concentrations are seen and renal involvement is manifested by nephrolithiasis and nephrocalcinosis. A new technology, recently approved for clinical use in the United States and Europe, is likely to become more widely available because it is an adaptation of the lumbar spine DXA image. Trabecular bone score (TBS) is a gray-level textural analysis that provides an indirect index of trabecular microarchitecture. Newer technologies, such as high-resolution peripheral quantitative computed tomography (HR-pQCT), have provided further understanding of the microstructural skeletal features in PHPT.

A doença óssea no hiperparatiroidismo primário grave é representada pela osteíte fibrosa cística (OFC). Dor óssea, deformidades esqueléticas e fraturas patológicas são achados comuns na OFC. A densidade mineral óssea está, usualmente, extremamente diminuída na OFC, mas é reversível após a cura cirúrgica. Os sinais e sintomas da doença óssea grave incluem dor óssea, fraturas patológicas e fraqueza muscular proximal com hiper-reflexia. O comprometimento ósseo é tipicamente caracterizado pela aparência em “sal-e-pimenta” nos ossos do crânio, erosões ósseas e reabsorção das falanges, tumores marrons e cistos. Na radiografia, observam-se desmineralização difusa e fraturas patológicas especialmente nos ossos longos das extremidades. No hiperparatiroidismo primário (HPTP) sintomático grave, as concentrações séricas de cálcio e PTH estão usualmente bem elevadas e o comprometimento renal se caracteriza pela presença de urolitíase e nefrocalcinose. Uma nova tecnologia, recentemente aprovada para uso clínico nos Estados Unidos e na Europa, torna-se provável se difundir rapidamente, pois utiliza as imagens geradas pela densitometria DXA. O escore trabecular ósseo (TBS), obtido por meio da análise do nível da textura cinza das imagens dos corpos vertebrais, fornece informações indiretas sobre a microarquitetura trabecular. Novos métodos, como a tomografia de alta resolução quantitativa periférica computadorizada (HRpqCT), têm proporcionado conhecimentos adicionais sobre os achados da microarquitetura esquelética no HPTP.