Clinical profile of primary hyperparathyroidism in Northeast India: a single centre experience

Background: A retrospective study of the presentation of primary hyperparathyroidism was done at a tertiary care centre in northeast India and was compared with variable features in other parts in India and worldwide.Methods: The clinical presentation, biochemical parameters, radiological and histopathology findings of 27 subjects of primary hyperparathyroidism who presented to us over a period of 5 years were retrospectively analysed. Chi-square test, student t test and 'one way ANOVA' were used to compare different variables. Statistical significance was set at p<0.05.Results: The age distribution ranged from as young as 13 years to 72 years (39±16.7). The male:female ratio was 1:1.25. The duration of symptoms at presentation ranged from 2 to 72 months (21.7±20.3). The most common presentation was bone pain in 59.2% of cases, followed by proximal myopathy (48.1%), fatigue (44.4%), abdominal pain (44.4%), constipation (11.1%), hypertension (18.5%), palpable neck swelling (22.2%), limb deformity (22.2%) and fracture (14.8%). The mean serum calcium was 12.2±0.87mg/dl. Parathyroid adenoma was localized radiologically in all patients and single adenoma was the most common cause in 96.3%. Left inferior parathyroid adenoma was the most common site of involvement in 51.8%.Conclusions: Hyperparathyroidism at our centre in northeast India has a classic symptomatic presentation with severe bone and renal involvement and younger age at diagnosis, and equal gender distribution.

Mineral and bone metabolism disorder in an adenine-induced rat model of chronic kidney disease / 中国比较医学杂志

Objective To investigate the characteristic changes of biochemical markers of mineral metabolism, vascular calcification, and renal osteodystrophy in an adenine-induced rat model of chronic kidney disease (CKD). Methods A total of 20 male Sprague Dawley rats (SD rats) were randomly divided into two groups: the normal group fed with a diet without adenine, and the CKD group fed with an adenine-containing diet (7. 5 g/kg) for the first 4 weeks and then a diet without adenine for the following 2 weeks. At the end of the 2nd week, serum biochemical markers were detected. At the end of the 6th week, the SD rats were sacrificed and serum biochemical markers were detected once again. The aortas were collected for pathological examination and detection of vascular calcium and phosphorus contents. Femurs and the fifth lumbar vertebrae were taken for bone mineral density (BMD) measurement and bone histomorphometric analysis. Results At the end of the 2nd and 6th weeks, compared with the normal control group, the levels of serum creatinine, urea nitrogen, phosphorus and parathyroid hormone (PTH) in the CKD group were significantly increased (P＜0. 05 or P＜ 0. 01), and the level of serum calcium was significantly decreased (P＜ 0. 05 or P＜ 0. 01). Medial layer vascular calcification of the aorta occurred in 50％ of the rats in the CKD group, but was not observed in the normal control group. Vascular calcium and phosphorus contents were significantly higher in the CKD group compared with the normal control group (P＜ 0. 05). The BMD of total femur, cortical and trabecular bone tissues of the femur, and the fifth lumbar vertebra was significantly decreased in the CKD group (P＜ 0. 05 or P＜ 0. 01). The histomorphometric analysis showed that both bone resorption and bone formation of the trabecular bone in the CKD group were increased, indicating a high bone turnover status. The volumes of both trabecular and cortical bones of rats in the CKD group were significantly lower than that of the normal control group (P ＜ 0. 05 or P ＜ 0. 01). However, the trabecular bone mineralization was not significantly different between the two groups. Conclusions The adenine-induced rat model of chronic kidney disease (CKD) established in this study shows reduced serum calcium and increased serum phosphorus and PTH, and medial layer vascular calcification of the aorta. With respect to renal osteodystrophy, this model shows a high trabecular bone turnover, normal trabecular bone mineralization, and low bone volume of cortical and trabecular bone, which meets the characteristics of osteitis fibrosa. This model may become a useful tool for future study of chronic kidney disease-mineral and bone disorder (CKD-MBD).

Subchondral Bone Restoration of Supra-acetabular Brown Tumor Secondary to Parathyroid Carcinoma: A Case Report / 대한고관절학회지

The causes of osteolytic lesions found in radiological examinations are not quite certain. Therefore, to determine the appropriate treatment method, various approaches and analyzes are required to find the real cause. Hyperparathyroidism is one of the diseases which forms osteolytic bone lesions so-called brown tumor. A 55-year-old woman who had painful osteolytic bone lesions in both hip joint areas was diagnosed as parathyroid carcinoma after serial work-up. She underwent parathyroidectomy and follow-up imaging showed a decrease in brown tumor size and bone consolidation in the subchondral bone destruction area. Proper evaluation of osteolytic bone lesions helps to avoid unnecessary operative treatments and the first choice for the treatment of osteolytic bone lesions caused by parathyroid carcinoma is parathyroidectomy.

Tumores pardos mimetizando enfermedad metastásica ósea como presentación inicial de un carcinoma paratiroideo: A propósito de un caso / Brown tumors mimetizing bone metastasic disease as an initial presentation of a parathyroid carcinoma: About a case

Objetivo: Describir caso inusual de paciente con tumor pardo, manifestación poco frecuente del hiperparatiroidismo, como presentación inicial de carcinoma de paratiroides. Caso Clínico: Paciente masculino de 25 años, inicia enfermedad actual en el 2011, caracterizada por dolor en encía inferior de hemiarcada izquierda, acompañada de tumoración en región maxilar inferior ipsilateral. Se evidencia progresión de dicha lesión y aparición de nueva tumoración en maxilar superior derecho, además de dolores óseos, limitación a la movilización y fracturas patológicas en húmeros. En abril de 2015 presenta disartria, dificultad en la alimentación por progresión de la tumoración en la cavidad oral, además de sangrado en el sitio de la lesión, por lo que es hospitalizado. Examen físico: lesión tumoral en maxilar superior e inferior que deforma la arcada dentaria, con sangrado activo que imposibilita la oclusión completa de boca, con asimetría facial. Extremidades: asimetría en ambos húmeros. Paraclínicos: PTH: 990 pg/mL, calcio: 13 mg/dL, fosfatasa alcalina: 300 UI/L. Se realiza paratiroidectomía y lobectomía tiroidea izquierda. Biopsia: carcinoma paratiroideo. A los meses se evidencia mejoría del dolor óseo y del tamaño de las lesiones. Conclusión: El carcinoma de paratiroides es una neoplasia rara, la prevalencia es menos de 1% de los casos de hiperparatiroidismo, y la incidencia es de 0.015 por 10000 casos. El tumor pardo es una forma infrecuente de manifestación de un hiperparatiroidismo, reflejando un desafío en el diagnóstico diferencial de una enfermedad metastásica ósea, y más aún cuando el carcinoma paratiroideo generalmente al momento del diagnóstico ya presenta metástasis a distancia.

Objective: To describe the unusual case of a patient with brown tumor, rare manifestation of hyperparathyroidism, as initial presentation of a parathyroid carcinoma. Case Report: Two years later evidence progression of the lesion, also appearance of new tumor in the right upper jaw, in addition to bone pain, limitation mobilization and pathological fractures in the humerus. In April 2015 the patient presents dysarthria and difficulty in feeding due to progression of the tumor in the oral cavity, in addition to bleeding at the site of injury, so it is hospitalized. Physical examination: tumor lesion in upper and lower jaw, which deforms the dental arch, with active bleeding that precludes complete occlusion of the mouth, with facial asymmetry. Limbs: asymmetry in both humerus. Paraclinical: PTH: 990 pg/mL, calcium: 13 mg/dL, alkaline phosphatase: 300 IU/L. Parathyroidectomy and left thyroid lobectomy was performed. Biopsy: parathyroid carcinoma. Months later, improvement in bone pain and lesion size was evident. Conclusions: Parathyroid carcinoma is a rare neoplasm, the prevalence is less than 1% of cases of hyperparathyroidism, and the incidence is 0.015 per 10,000 cases. The brown tumor is a rare form of manifestation of hyperparathyroidism, reflecting a challenge in the differential diagnosis of a metastatic bone disease, and even more when the parathyroid carcinoma usually at the time of diagnosis already presents distant metastases.

Pathologic Fracture of the Femur in Brown Tumor Induced in Parathyroid Carcinoma: A Case Report / 대한고관절학회지

Brown tumor refers to a change of skeletones that develops as a complication of hyperparathyroidism. As osteoclast is activated to stimulate reabsorption and fibrosis of bone, it causes a cystic change of the bone. Parathyroid carcinoma is being reported as a tumor that induces primary hyperparathyroidism. It causes excessive secretion of the parathyroid hormone and increases the blood parathyroid hormone and calcium. Bone deformation due to brown tumor is known to be naturally recovered through the treatment for hyperparathyroidism. However, there is no clearly defined treatment for lesions that can induce pathological fractures developing in lower extremities. We experienced a case where brown tumor developed in the proximal femur of a 57-year-old female patient due to parathyroid carcinoma. In this case, spontaneous fracture occurred without any trauma, and it was cured by performing intramedullary nailing fixation and parathyroidectomy. We report the treatment results along with a literature review.

Brown Tumor of the Cervical Spines: A Case Report with Literature Review

To report a rare case of axis brown tumor and to review literature of cervical spine brown tumor. Brown tumor is a rare bone lesion, incidence less than 5% in primary hyperparathyroidism. It is more common in secondary hyperparathyroidism with up to 13% of cases. Brown tumor reactive lesion forms as a result of disturbed bone remodeling due to long standing increase in parathyroid hormones. Cervical spine involvement is extremely rare, can be confused with serous spine lesions. To date, only four cases of cervical spine involvement have been reported. Three were due to secondary hyperparathyroidism. Only one was reported to involve the axis and was due to secondary hyperparathyroidism. This is the first reported case of axis brown tumor due to primary hyperparathyroidism. A case report of brown tumor is presented. A literature review was conducted by a Medline search of reported cases of brown tumor, key words: brown tumor, osteoclastoma and cervical lesions. The resulting papers were reviewed and cervical spine cases were listed then classified according to the level, cause, and management. Only four previous cases involved the cervical spine. Three were caused by secondary hyperparathyroidism and one was by primary hyperparathyroidism which involved the C6. Our case was the first case of C2 involvement of primary hyperparathyroidism and it was managed conservatively. Brown tumor, a rare spinal tumor that presents with high PTH and giant cells, requires a high level of suspicion.

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.

A Case of Parathyroid Carcinoma Presenting as Brown Tumors / 대한이비인후과학회지

Carcinoma of the parathyroid gland has been reported as a rare disease occurring in 0.5 to 4% of patients with the primary hyperparathyroidism. Brown tumor is characterized as the terminal stage of remodeling processes during the primary or secondary hyperparathyroidism. Brown tumor induced by the primary hyperparathyroidism can commonly occur in the ribs, clavicle, and pelvic bone. We experienced a case of a 29-year old female patient, who had parathyroid carcinoma leading to primary hyperparathyroidism, followed by multiple Brown tumor. We present this case of parathyroid carcinoma with Brown tumors with a brief review of literature.

Osteitis Fibrosa Cystica Mistaken for Malignant Disease

A 65-year-old man with back pain had plain radiographs that showed multiple osteolytic bone lesions of the pelvis, femur and L-spine; an magnetic resonance imaging scan of the L-spine showed extensive bony resorption with a posterior epidural mass involving the L1 spinous process; these findings suggested multiple myeloma or bony metastasis. However, all serology testing was negative. The parathyroid hormone and serum calcium levels were found to be abnormally elevated. A fine needle aspiration biopsy suggested that the L-spine lesion was consistent with the diagnosis of osteitis fibrosa cystica. A pathological fracture of the spine compressed the spinal cord, and surgical intervention was required. The neck computed tomography and Tc-99m sestamibi scan showed a solitary parathyroid mass. A minimally invasive parathyroidectomy using intraoperative parathyroid hormone monitoring was performed and two enlarged parathyroid glands identified. This case illustrates the importance of the consideration of a rare brown tumor associated with primary hyperparathyroidism in patients with the bone lesions suggestive of a malignancy.

Tumores pardos múltiples en un paciente con hiperparatiroidismo primario / Multiple brown tumors in primary hyperparathyroidism: report of one case

Brown tumors are an uncommon manifestation of primary and secondary hyperparathyroidism. We report a 38 years old male consulting for generalized bone pain and prostration caused by multiple osteolytic lesions. Diagnostic work up disclosed a primary hyperparathyroidism secondary to a right parathyroid adenoma. The patient was subjected to a parathyroidectomy. After one year of follow up, symptoms have decreased considerably, laboratory parameters are normal and bone lesions are disappearing.

Primary hyperparathyroidism in children and adolescents.

Objective. Primary hyperparathyroidism (PHPT) in children and adolescents is a rare condition. Awareness should improve in order to lower threshold for screening and allow intervention before serious and permanent sequelac occur. Methods. A retrospective analysis of 15 children and adolescents with PHPT (age <20 yr) seen in our clinic between 1993 and 2006. Results. Mean age of patients was 17.73 yr (Range - 13-20, Male-3: Female-12). Average duration of symptoms was 18.87 (range: 0-48) mo. Clinical features at presentation included bone pain (86.67%), proximal myopathy (46.67%), bony deformities (53.33%), fractures (60%), palpable osteitis fibrosa cystica (33.3%), renal calculi (40%), palpable neck swelling (13.3%) and acute pancreatitis (6.67%). None had positive family history or features suggestive of multiple endocrine neoplasia (MEN). After biochemical confirmation, tumor was localised in all prior to surgery. Histopathology confirmed adenoma in all cases. Post-operative hungry bone syndrome occurred in 33.3%. Conclusion. PHPT is more common in females. Presentation of the disease is similar to their adult counterparts except for more severe bone disease and less severe renal disease. MEN and familial non-MEN PHPT do not constitute a major cause of pediatric PHPT as against to worldwide data. The incidence of hyperplasia as a cause of PHPT is rare in our pediatric population.

Multiple brown tumors of the jaws in primary hyperparathyroidism / 대한구강악안면방사선학회지

Brown tumor is usually diagnosed at the terminal stage of hyperparathyroidism. Diagnosis of this tumor is confirmed by endocrinologic investigations along with clinical and radiographic examination. Radiographical differential diagnosis of this tumor includes central giant cell granuloma, aneurysmal bone cyst, metastatic tumor, multiple myeloma, and Paget disease. This report presents a rare case of multiple brown tumors occurring at the maxilla and mandible, which was initially misdiagnosed as central giant cell granuloma. Plain radiographs demonstrated multiple well-defined multilocular radiolucency. CT images showed soft tissue mass with low attenuated lesions, perforation of the lingual cortical plate, and a heterogeneous mass at the right thyroid lobe. These findings were consistent with parathyroid adenoma. The patient had hypercalcemia, hypophosphatemia, and elevated alkaline phosphatase level. Surgical excision of the tumor was performed. No recurrence was observed during a 28-month follow-up.

Manifestaciones clínicas inusuales del hiperparatiroidismo primario / Unsual clinical signs of primary hyperparathroidism

Introducción: Un creciente número de pacientes con hiperparatiroidismo primario son diagnosticados en la ausencia de síntomas, gracias a la determinación rutinaria de calcio sérico. Sin embargo, en algunos países las manifestaciones típicas continúan dominando la presentación del hiperparatiroidismo primario. Métodos: Estudiamos retrospectivamente las manifestaciones clínicas y bioquímicas de 47 pacientes consecutivos con hiperparatiroidismo primario, tratados con paratiroidectomía entre octubre de 1993 y junio de 2005. Resultados: La edad media fue de 51.3 años. Se incluyeron 40 mujeres (85%) y siete varones (15%). El 63% de los pacientes fue referido por la sospecha de neoplasia maligna. En 78% de los casos se identificaron lesiones radiológicas compatibles con osteítis fibrosa quística, resorción subperóstica, lesiones líticas múltiples, osteopenia y osteoporosis. La presencia de fractura en terreno patológico se observó en 19.1%. Quince pacientes (32%) tenían tumores pardos, con localización más frecuentemente en la mandíbula y la maxila. Conclusiones: En esta serie, la osteítis fibrosa quística sintomática y la severa disminución de la densidad mineral ósea fueron las manifestaciones dominantes del hiperparatiroidismo primario. En su mayoría, los pacientes fueron referidos a una unidad oncológica para su tratamiento debido a la sospecha de alguna neoplasia maligna. Una adecuada evaluación clínica, bioquímica, radiológica e histológica es necesaria para establecer el posible diagnóstico de hiperparatiroidismo.

BACKGROUND: A growing number of patients with primary hyperparathyroidism (PHPT) are diagnosed in the absence of symptoms following routine biochemical screening. However, in some countries, overt manifestations and osteitis fibrosa cystica (OFC) still dominate the clinical profile of PHPT patients. METHODS: We retrospectively studied clinical and biochemical manifestations of 47 consecutive patients with primary hyperparathyroidism who were treated with parathyroidectomy from October 1993 to June 2005. RESULTS: Mean age was of 51.3 years. Our sample included 40 women (85%) and 7 men (15%). Seventy eight percent of cases had radiological features of OFC, namely subperiosteal bone resorption, cortical cysts and osteopenia. Pathological fracture occurred in nine patients (19.1%). Fifteen (32%) patients had clinically evident bony deformities or brown tumors mostly located in the mandible and maxilla. CONCLUSIONS: Our results indicate that symptomatic osteitis fibrosa cystica and severe decrease of bone mineral density were the primary manifestations of primary hyperparathyroidism. Most patients were referred to an oncology hospital for treatment due to a suspected malignant neoplasm. A comprehensive clinical evaluation with biochemical markers, imaging studies and histological results is needed to establish a possible diagnosis of primary hyperparathyroidism.

Prevalence of radiological findings among cases of severe secondary hyperparathyroidism / Prevalência de achados radiológicos em pacientes com grave hiperparatireoidismo secundário

CONTEXT AND OBJECTIVE: Patients with end stage renal disease (ESRD) and secondary hyperparathyroidism (HPT2) are prone to develop heterotopic calcifications and severe bone disease. Determination of the sites most commonly affected would decrease costs and patients' exposure to X-ray radiation. The aim here was to determine which skeletal sites produce most radiographic findings, in order to evaluate hemodialysis patients with HPT2, and to describe the most prevalent radiographic findings. DESIGN AND SETTING: This study was cross-sectional, conducted in one center, the Hospital Universitário Clementino Fraga Filho (HUCFF), in Rio de Janeiro, Brazil. METHODS: Whole-body radiographs were obtained from 73 chronic hemodialysis patients with indications for parathyroidectomy due to severe HPT2. The regions studied were the skull, hands, wrists, clavicles, thoracic and lumbar column, long bones and pelvis. All the radiographs were analyzed by the same two radiologists, with great experience in bone disease interpretation. RESULTS: The most common abnormality was subperiosteal bone resorption, mostly at the phalanges and distal clavicles (94 percent of patients, each). "Rugger jersey spine" sign was found in 27 percent. Pathological fractures and deformities were seen in 27 percent and 33 percent, respectively. Calcifications were presented in 80 percent, mostly at the forearm fistula (42 percent), abdominal aorta and lower limb arteries (35 percent each). Brown tumors were present in 37 percent of the patients, mostly on the face and lower limbs (9 percent each). CONCLUSION: The greatest prevalence of bone findings were found on radiographs of the hands, wrists, lateral view of the thoracic and lumbar columns and femurs. The most prevalent findings were bone resorption and ectopic calcifications.

CONTEXTO E OBJETIVO: Pacientes com hiperparatireoidismo secundário (HPT2) à insuficiência renal crônica são propensos a desenvolver calcificações ectópicas e grave doença óssea. A determinação dos sítios mais revelantes pode diminuir o custo e a exposição do paciente a radiação desnecessária. O objetivo foi determinar quais locais radiológicos apresentam mais achados radiográficos para avaliar o HPT2 em pacientes em hemodiálise, assim como os achados mais prevalentes. TIPO DE ESTUDO E LOCAL: Estudo transversal, realizado no Hospital Universitário Clementino Fraga Filho (HUCFF), no Rio de Janeiro, Brasil. MÉTODOS: Radiografias de corpo inteiro foram obtidas de 73 pacientes em hemodiálise crônica que tiveram indicação de paratireoidectomia devido a HPT2 grave. As regiões estudadas foram crânio, mãos e punhos, clavículas, coluna torácica e lombar, ossos longos e pélvis. Todas as imagens foram analisadas pelos mesmos dois radiologistas, com grande experiência na interpretação de doenças do osso. RESULTADOS: A alteração mais comum foi reabsorção óssea subperiosteal, principalmente nas falanges distais e clavículas (ambos em 94 por cento de pacientes). Sinal de "rugger jersey" foi descoberto em 27 por cento. Fratura patológica e deformidades foram visualizadas em 27 por cento e 33 por cento, respectivamente. As calcificações foram encontradas em 80 por cento dos pacientes, principalmente na fístula de antebraço (42 por cento), aorta abdominal e artérias dos membros inferiores (ambos 35 por cento). Tumores marrons estavam presentes em 37 por cento dos pacientes, principalmente na face e nos membros inferiores (ambos 9 por cento). CONCLUSÃO: As radiografias com achados mais prevalentes foram mãos e punhos, radiografia lateral da coluna torácica e lombar e fêmur. Os achados mais prevalentes foram reabsorção óssea e calcificação ectópica.

Osteíte fibrosa cística causando paraplegia crural / Crural paraplegia for osteitis fibrosa cystica

A osteíte fibrosa cística é uma apresentação avançada do hiperparatireoidismo e pode acometer, mais raramente, a coluna vertebral. No presente relato, a paciente evoluiu de um quadro de dor localizada para paraplegia por compressão medular. Destaca-se a importância de investigação e abordagem precoces dessa condição, a fim de evitar complicações irreversíveis.

Osteitis fibrosa cystica is an advanced consequence of hiperparathyroidism and, eventually, it is situated in spine. In this report, the patient showed a localized pain which quickly evaluated to paraplegia caused by spinal cord compression. Early investigation and intervention in this clinical condition are important in order to prevent irreversible complications.

Tumor pardo como manifestación inicial de hiperparatiroidismo primario / Brown tumor as the initial manifestation of primary hyperparathyroidism

El tumor pardo es una forma localizada de osteítis fibrosa quística, parte del compromiso óseo por hiperparatiroidismo. Como primera expresión de hiperparatiroidismo es infrecuente, debido a que actualmente éste se diagnostica en estadios asintomáticos o mínimamente sintomáticos. Presentamos el caso de una paciente con un tumor pardo localizado en el maxilar superior izquierdo, como primera manifestación de hiperparatiroidismo primario causado por un adenoma paratiroideo. Posterior a la realización de una paratiroidectomía el tumor evolucionó con franca regresión, sin necesidad de ningún otro procedimiento quirúrgico local.

Brown tumor is a localized form of osteitis fibrosa cystica, being part of the hyperparathyroid bone disease. It rarely is the first manifestation of hyperparathyroidism, since nowadays, the diagnosis is made at an asymptomatic or minimally symptomatic stage. We present a case of a left superior maxillar brown tumor as the first manifestation of primary hyperparathyroidism due to a parathyroid adenoma. A parathyroidectomy was performed, and there was a regression of the bone lesion, without the need of performing other local surgical procedures.

Múltiples tumores pardos maxilofaciales como manifestación de hiperparatiroidismo primario / Multiple maxillofacial brown tumors as primary hyperparathyroidism manifestation

Objetivo: Con el fin de analizar el diagnóstico diferencial de las lesiones óseas con células gigantes en los huesos faciales, presentamos un caso con hiperparatiroidismo primario no diagnosticado previamente, que presentó múltiples tumores pardos maxilofaciales como primera manifestación clínica de la enfermedad. Caso clínico: Mujer de 70 años de edad con tumor en el arco anterior de la mandíbula de un año de evolución. Una biopsia confirmó la presencia de una lesión con células gigantes. Radiológicamente se corroboró la presencia de otras dos lesiones líticas en la región maxilofacial. Durante la evaluación bioquímica previa a la cirugía se consideró la posibilidad de hiperparatiroidismo. Por tomografía computarizada se localizó tumor de paratiroides en una posición atípica. La resección quirúrgica confirmó adenoma de paratiroides. La paciente cursó con hipocalcemia sintomática, siendo manejada con suplementos de calcio y calcitriol. Al cuarto mes de la cirugía, persistía con cifras normales de calcio sérico y el tumor mandibular se había reducido parcialmente. Conclusiones: La detección de una lesión ósea con células gigantes en la región maxilofacial es un elemento diagnóstico primordial puesto que varias entidades, entre ellas el tumor pardo del hiperparatiroidismo, pueden tener una imagen histológica similar. Sólo una evaluación clínica, radiológica y bioquímica sistemática puede permitir un diagnóstico definitivo. La presencia de múltiples tumores pardos maxilofaciales simultáneos en el hiperparatiroidismo primario es poco común, y en raras ocasiones puede ser el primer signo de la enfermedad.

OBJECTIVE: In order to analyze the differential diagnosis of giant-cell lesion in facial bones, we present a case of a patient without a previously diagnosed primary hyperparathyroidism that displayed multiple maxillofacial brown tumors as the initial clinical manifestation of the disease. CASE DESCRIPTION: A 70 year-old female with amandible tumor and one year of disease progression. Tumor biopsy confirmed the presence of a giant-cell lesion. Radiologically, we confirmed the presence of another two lytic lesions in the maxillofacial region. During biochemical evaluation prior to surgery, the possibility of hyperparathyroidism was considered. Using computed tomography, we noted a parathyroid tumor in an atypical location. Surgical resection confirmed the presence of an adenoma. Postoperatively, the patient developed symptomatic hypocalcemia and was managed with calcium supplementation in addition to calcitriol. At 4 months after surgery mandibular swelling had regressed partially and serum calcium levels returned to normal levels. CONCLUSION: The detection of giant-cell bone lesions in the maxillofacial region is a strategic diagnostic finding as several entities, among these brown tumor hyperparathyroidism can display similar histologic imaging findings. Only systematic clinical, radiologic, and biochemical evaluation can allow for a definitive diagnosis. The presence of multiple simultaneous maxillofacial brown tumors in primary hyperparathyroidism is an infrequent ocurrence, and only on rare occasions can this be the first sign of the disease.

A Case of Cystic Parathyroid Adenoma Presenting as Severe Bony Lesion / 대한내분비학회지

A cystic parathyroid adenoma is rare. A case of primary hyperparathyroidism, with the cystic formation of a parathyroid adenoma and a severe bony lesion, is reported. A 52-year-old male was admitted due to pain in both hips and for evaluation of hypercalcemia. The plasma level of the intact parathyroid hormone(iPTH) was elevated to 1424pg/mL. Ultrasonography and the computed tomography revealed a parathyroid cyst on the left thyroid lower pole. Parathyroid scintigraphy detected a parathyroid adenoma. A radiograph showed a subperiosteal bone resorption on the phalanges, and a brown tumor(osteitis fibrosa cystica) on the femur shaft was noted. A surgical excision of the parathyroid adenoma was performed. The PTH level in the cystic fluid was increased. A histological examination confirmed a cystic parathyroid adenoma. The PTH level was normalized after the operation.