Tumor pardo del paladar, presentación atípica de hiperparatiroidismo primario en adolescente: buena evolución y normalización de la masa ósea tras paratiroidectomía / Brown tumor of the palate, atypical presentation of primary hyperparathyroidism in an adolescent: good evolution and bone mass normalization after parathyroidectomy

El hiperparatiroidismo primario (HPTP) es poco frecuente en niños y adolescentes. Hay escasos datos para el manejo de estos pacientes en pediatría. Las glándulas paratiroideas son glándulas endocrinas que secretan hormona paratiroidea (PTH) y regulan el metabolismo del calcio y del fósforo. La sobreexpresión de PTH se llama hiperparatiroidismo, que se clasifica en primario, secundario y terciario. En los adolescentes, 80 a 92% de los hiperparatiroidismos primarios se deben a adenoma paratiroideo. Presentamos el caso clínico de una adolescente con una primera manifestación atípica de HPTP, la presencia de un tumor pardo del paladar, presentación rara de adenoma paratiroideo, acompañado de hipercalcemia, marcada elevación de PTH y varias lesiones óseas. (AU)

Primary hyperparathyroidism (PHPT) in children and adolescents is uncommon. Guidelines for management in pediatric patients are limited. Parathyroid glands are endocrine glands that secrete parathyroid hormone (PTH) and regulate calciumphosphate metabolism. The overexpression of PTH is called hyperparathyroidism, and is classified as primary, secondary, and tertiary. In adolescents, 80 to 92% of PHPT cases are due to a parathyroid adenoma. We present here a case report of an adolescent with a brown tumor of the palate as the first manifestation of the disease, atypical and rare presentation of parathyroid adenoma in an adolescent. She had hypercalcemia, marked elevation of PTH and bone lesions. (AU)

Tumor de células gigantes (osteoclastoma) multicentrico, diagnóstico incidental de adenoma paratiroideo reporte de caso / Multicenter giant cell tumor (osteoclastoma), incidental diagnosis of parathyroid adenoma case report

Los tumores de células gigantes (TDG) llamados también osteoclastomas o tumores pardos cuando se hallan dentro la esfera endocrinológica, son uno de los tumores menos frecuentes, más controversial y menos predecible en su comportamiento. Se producen como consecuencia del exceso de la actividad osteoclastica, como ocurre en el caso del hiperparatiroidismo, mismo que es un desorden endocrino común, por lo general asintomático y diagnosticado por el hallazgo fortuito de hipercalcemia. El diagnóstico de los osteoclastomas suele ser un reto, el alto índice de sospecha es esencial y la biopsia es el estándar de oro para el diagnóstico. Presentamos el caso de un hombre de 42 años quien presentó fracturas patológicas de radio derecho y tibia izquierda, gammagrama óseo con Tc ­ 99m con múltiples lesiones óseas , hormona paratiroidea (PTH) elevada, hipercalcemia, gammagrama de paratiroides con MIBI con presencia de adenoma paratiroideo, la biopsia de las lesiones óseas con presencia de células gigantes multinucleadas correspondientes a osteoclastomas; se llevó a cabo paratiroidectomia y el examen histopatológico confirmó la presencia de un adenoma paratiroideo.

Giant cell tumors (TDG), also called osteoclastomas or brown tumors when they are within the endocrinological sphere, are one of the least frequent, most controversial and least predictable tumors in their behavior. They occur as a consequence of excess osteoclastic activity, as occurs in the case of hyperparathyroidism, which is a common endocrine disorder, generally asymptomatic and diagnosed by the fortuitous finding of hypercalcemia. Diagnosing osteoclastomas is usually challenging, the high index of suspicion is essential, and biopsy is the gold standard for diagnosis. We present the case of a 42-year-old man who presented pathological fractures of the right radius and left tibia, a bone scan with Tc-99m with multiple bone lesions, elevated parathyroid hormone (PTH), hypercalcemia, parathyroid scan with MIBI with the presence of parathyroid adenoma, the biopsy of the bone lesions with the presence of multinucleated giant cells corresponding to osteoclastomas; parathyroidectomy was performed and histopathological examination confirmed the presence of a parathyroid adenoma.

Repeated epistaxis caused by primary hyperparathyroidism with multiple brown tumors: One case report / 中华内分泌代谢杂志

To report the clinical, imaging and pathological feature of a case of primary hyperparathyroidism(PHPT) with multiple brown tumors whose complain was repeated epistaxis, and to conduct a retrospective analysis with relevant literatures. A postmenopausal middle-aged woman, with a history of joint pain, fracture, and kidney stone which were neglected due to the lack of specificity of symptoms for years, went to hospital due to repeated epistaxis. Laboratory results showed high blood calcium, hypophosphatemia, alkaline phosphatase(ALP), and parathyroid hormone(PTH) levels, as well as increased urinary calcium. Imaging examination showed systemically multiple brown tumors and extensive osteoporosis. Meanwhile an abnormal mass was found behind the right thyroid. Taking all findings into account, the diagnosis was made with PHPT, and the histopathology further confirmed that the lesion was a parathyroid adenoma. This case seems to be a rare case of multiple brown tumor caused by parathyroid adenoma, which compressed and destructed nasal blood vessels, thus induced a repeated nasal discharge. After resection of parathyroid adenoma, the patient experienced a transient hypocalcemia and PTH level elevation. After 5-month follow up, part of the brown tumors spontaneously regressed, bone density increased, and her clinical symptoms were mostly relieved.

Lesiones óseas multicéntricas por trastorno paratiroideo: Reporte de un caso clínico / Multicentric Bone Lesions Due to Parathyroid Disease: A Case Report

Resumen Introducción: El hiperparatiroidismo (Hpt) es un desorden endócrino que crea condiciones de hipersecreción de la hormona paratiroidea, propicias para el asentamiento de tumores en el tejido óseo. Si bien no es frecuente, los huesos maxilares, pueden estar involucrados en la patogenia de estas lesiones. Pacientes y métodos: En este trabajo, se presenta un caso clínico de una paciente femenina de 56 años de edad, con lesiones óseas multicéntricas maxilares y extramaxilares a predominio de células gigantes, vinculado a un hiperparatiroidismo. Discusión: El diagnóstico de un Tumor Pardo del Hiperparatiroidismo (TPH) es difícil en la clínica estomatológica, donde la metodología de diagnóstico debe reunir criterios serológicos, clínicos e histopatológicos. La multifocalidad de las lesiones, hacen a este caso un verdadero síndrome endócrino, siendo éste el aspecto más importante. Esta entidad, de incumbencia en la Endocrinología y en la Medicina Bucal, debe ser tratada fundamentalmente de forma interdisciplinar para lograr un tratamiento exitoso.

Abstract Introduction: Hyperparathyroidism is an endocrine disorder that could create parathyroid hormone hypersecretion conditions, propitious for bone tumors development. Although is not frequent, maxillary bones may be involved in the pathogenesis of these lesions. PATIENTS AND METHODS: In this case, a 56-year-old female is reported. An Hyperparathyroidism was diagnosed due to multiple maxillary and extra-maxillary multicentric bone lesions, with a predominance of giant cells, linked to her endocrine disorder. Discussion: The diagnosis of a Hyperparathyroidism Brown Tumor must meet serological, clinical and histopathological criteria. The multifocality of the lesions is the most remarkable clinical aspect in this case. For an adequate treatment of this entity is necessary an interdisciplinary and coordinate work between different areas of healthcare physicians and Oral Medicine specialist.

Múltiples tumores pardos en una paciente con hiperparatiroidismo secundario grave / Brown tumors in a patient with severe secondary hyperparathyroidism

Resumen El hiperparatiroidismo secundario es una complicación frecuente de la insuficiencia renal crónica. Los tumores pardos son una variante de osteítis fibrosa quística raramente reportados en pacientes con insuficiencia renal terminal como consecuencia del incremento en la actividad osteoclástica.

Abstract Secondary hyperparathyroidism is a common complication of chronic renal failure. Brown tumors are a variant of cystic fibrous osteitis rarely reported in patients with end stage renal failure as a result of increased osteoclast activity.

Tumor marrom em maxila associado ao hiperparatireoidismo secundário: relato de caso clínico / Brown tumour of the jaws associated with secondary hiperparathyroidism: case report

O tumor marrom é uma lesão intraóssea benigna focal associada ao hiperparatireoidismo. Ocorre preferencialmente em ossos longos, costelas e pelve, porém, eventualmente pode acometer maxila e mandíbula. O prognóstico está associado à causa e ao tipo de hiperparatireoidismo, com isso é de grande importância o diagnóstico precoce para que seja estabelecido o quanto antes a terapêutica adequada. Apesar destas lesões serem raras nos ossos gnáticos e quando presentes estarem associadas principalmente ao hi per pa ratireoidismo primário e em mulheres com mais de 50 anos de idade, propusemos relatar um caso de tumor marrom secundário do hiperparatireoidismo em paciente do gênero masculino e de 36 anos de idade.

The brown tumor is a focal benign intraosseous lesion associated with hyperparathyroidism. Preferably occurs in long bones, ribs and pelvis, however, may eventually affect the jaws. The prognosis is associated with the cause and type of hyperparathyroidism, with that It is very important for an early diagnosis so it can soon be established an appropriate therapy. Although these are rare injuries on the gnathic bones and when present are mainly associated with primary hyperparathyroidism and in women over 50 years old, we proposed to report a case of secondary hyperparathyroidism brown tumor in a 36 years old male patient.

Brown Tumor of the Thoracic Spine: First Manifestation of Primary Hyperparathyroidism

Brown tumors also called as osteoclastomas, are rare nonneoplastic lesions that arise in the setting of primary or secondary hyperparathyroidism. Parathyroid adenomas or hyperplasia constitute the major Brown tumor source in primary hyperparathyroidism while chronic renal failure is the leading cause in secondary hyperparathyroidism. Most of the patients with the diagnosis of primary hyperparathyroidism present with kidney stones or isolated hypercalcemia. However, nearly one third of patients are asymptomatic and hypercalcemia is found incidentally. Skeletal involvement such as generalized osteopenia, bone resorption, bone cysts and Brown tumors are seen on the late phase of hyperparathyroidism. The symptoms include axial pain, radiculopathy, myelopathy and myeloradiculopathy according to their locations. Plasmocytoma, lymphoma, giant cell tumors and metastates should be ruled out in the differential diagnosis of Brown tumors. Treatment of Brown tumors involve both the management of hyperparathyroidism and neural decompression. The authors report a very rare spinal Brown tumor case, arisen as the initial manifestation of primary hyperparathyroidism that leads to acute paraparesis.

Primary Hyperparathyroidism with Extensive Brown Tumors and Multiple Fractures in a 20-Year-Old Woman

A brown tumor is a benign fibrotic, erosive bony lesion caused by localized, rapid osteoclastic turnover, resulting from hyperparathyroidism. Although brown tumors are one of the most pathognomonic signs of primary hyperparathyroidism, they are rarely seen in clinical practice. In this report, we present a case of 20-year-old woman with recurrent fractures and bone pain. Plain digital radiographs of the affected bones revealed multiple erosive bone tumors, which were finally diagnosed as brown tumors associated with primary hyperparathyroidism due to a parathyroid adenoma. This case shows that multiple, and clinically severe form of brown tumors can even occur in young patients.

Brown Tumor of the Patella Caused by Primary Hyperparathyroidism: A Case Report

It has been reported that the common sites of brown tumors are the jaw, pelvis, ribs, femurs and clavicles. We report our experience in a case of brown tumor of the patella caused by primary hyperparathyroidism. An initial radiograph and CT showed an osteolytic lesion and MR images showed a mixed solid and multiloculated cystic tumor in the right patella. One month after the parathyroidectomy, rapid bone formation was observed on both radiographs and CT images.

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.

Tumor pardo multifocal como manifestación del hiperparatiroidismo primario por adenoma paratiroideo asociado a carcinoma papilar de tiroides / Brown tumor of the pelvis associated with hyperparathyroidism

Background: Brown tumors of bones are an uncommon manifestation of hyperparathyroidism. Case report: We report a 35 years old male presenting with pain and paresis of the left superior limb. Part of his humerus was excised due to a diagnosis of a giant cell tumor. He was admitted again to the hospital due to pelvic pain, malaise and constipation. A right cervical nodule was found. Laboratory evaluation confirmed the presence of a hyperparathyroidism. The biopsy of the pelvic lesion disclosed a brown tumor. The patient was subjected to a parathyroidectomy and the pathological study of the surgical piece showed a right parathyroid adenoma and a right thyroid papillary micro carcinoma. In the postoperative period the patient had a hungry bone syndrome, which was adequately treated.

Introducción: La paratohormona es una hormona encargada de la homeostasis del calcio, el hiperparatiroidismo es una patología con manifestaciones renales y óseas, el Tumor Pardo es una rara presentación de esta enfermedad. Caso clínico: Hombre de 35 años con dolor y paresia en extremidad superior izquierda, fue resecado parte del húmero por un diagnóstico de Tumor de Células Gigantes; reingresa con dolor pélvico derecho, malestar general, astenia y estreñimiento. Se descubre un nódulo cervical derecho e hipersensibilidad en la pelvis derecha. Los exámenes de laboratorio muestran hiperparatiroidismo; la biopsia de la lesión pélvica es diagnóstica de Tumor Pardo, encontrándose además una hipercaptación paratiroidea derecha. Operado, el diagnóstico histopatológico fue: Adenoma paratiroideo derecho y un micro carcinoma papilar tiroideo; en el post-operatorio desarrolló un Síndrome de Bone Hunger, el cual fue superado y dado de alta. Discusión y conclusiones: El Tumor Pardo no es una verdadera neoplasia; producido por intensa actividad osteoclástica, tiene características histológicas y radiológicas inespecíficas y su diagnóstico se realiza por datos clínicos y bioquímicos. El hiperparatiroidismo puede llevar a la formación de Tumores Pardos; se sugiere realizar estudios de la glándula tiroides en pacientes con hiperparatiroidismo.

Hiperparatiroidismo primario con tumor pardo mimetizando enfermedad ósea maligna

Objetivo: Los tumores pardos son una manifestación infrecuente del hiperparatiroidismo primario (HPTP). Se presenta la evaluación diagnóstica en una paciente con lesión ósea e imágenes osteolíticas extensas que confunden y orientan a tumor óseo metastásico. Caso clínico: Paciente femenina de 49 años de edad, con dolor lumbosacro irradiado a cresta ilíaca izquierda y limitación para la marcha de dos años de evolución, con episodios de pancreatitis aguda e hipertrigliceridemia; en el último episodio se le realiza TAC abdómino-pélvica, con hallazgo incidental de lesión sacro ilíaca izquierda, que sugiere tumor óseo; se realizan estudios de extensión en pesquisa de metástasis. La biopsia ósea muestra células gigantes con proliferación fibroblástica y formación de hueso reactivo, sugestivo de tumor de células gigantes, el USG tiroideo muestra imagen hipoecoica, hipervascularizada, USG renal con nefrolitiasis renal. Laboratorio: PTH 1250 pg/mL y otra en 986,7 pg/mL. Calcio 9,8 mg/mL, Fosfatasa alcalina 570 U/L, Marcadores tumorales negativos. La Rx de cráneo muestra imágenes líticas en región frontal en “sal y pimienta”. DXA: osteoporosis cortical, T-score antebrazo izquierdo -3,1. TAC de cadera: lesión osteolítica expansiva con irrupción de la cortical, de 7 x 3.5 cm en cresta ilíaca izquierda y múltiples imágenes osteolíticas sacroilíacas y vertebrales (L4-L5). El CT-PET con áreas múltiples de hipercaptación que sugieren infiltración ósea por enfermedad metastásica, sin captación en cuello. Se realiza Gammagrama 99mTc-MIBI que revela imagen sugestiva de adenoma de paratiroides en el lóbulo izquierdo. Se realiza extirpación del mismo. Conclusiones: Las lesiones óseas múltiples son enfocadas en el contexto metastásico y pueden confundir el diagnóstico de HPTP. Los tumores pardos son causa potencial de falsos positivos con CT/PET en la evaluación de pacientes con tumor primario desconocido o metástasis esqueléticas. Análisis básicos de metabolismo cálcico y gammagrama paratiroideo con 99mTc-MIBI, son herramientas diagnósticas en estos pacientes.

Objective: Brown tumors are an uncommon manifestation of Primary Hyperparathyroisim (PHPT). We provide diagnostic evaluation in a patient with bone lesion and extensive osteolytic images that suggesting metastatic bone tumor. Case report: Female patient 49 years old with lumbosacral pain radiating to the left iliac crest and walking limitation of two years of evolution with episodes of acute pancreatitis and hypertriglyceridemia; in the last episode, abdominal and pelvic CT images were performed, with incidental finding of left sacroiliac bone lesion, that suggests a bone tumor. Extension studies on screening for metastases were performed. Bone biopsy showed giant cells with fibroblastic proliferation and reactive bone formation, suggestive of giant cell tumors; thyroid USG showed hypoechoic image, hypervascularized; renal USG showed nephrolithiasis. Laboratory: PTH 1250 pg/mL and another at 986.7 pg/mL, calcium 9.8 mg/mL, alkaline phosphatase 570 U/L and tumor markers negatives. X ray of skull showed multiple lytic lesions in the frontal region, like “salt and pepper”. DXA: cortical osteoporosis, with a left forearm T-score of -3.1. Computed tomographic of pelvis showed expansive osteolytic lesion with cortical irruption of 7 cm x 3.5 cm in the left iliac crest and multiple osteolytic vertebral and sacroiliac images (L4-L5). The CT-PET showed multiple areas of increased uptake suggesting infiltration by metastatic bone disease, no neck pickup. A 99mTc-MIBI scintigraphy revealed an image suggestive of parathyroid adenoma in the left lobe, which is excised. Conclusion: Multiple bone lesions are focused as metastasis and may confuse the diagnosis of PHPT. Brown tumors are potential causes of false positives with CT / PET in the evaluation of patients with unknown primary tumor or skeletal metastases. Basic analysis of calcium metabolism and parathyroid scintigraphy 99mTc.MIBI are diagnostic tools in these patients.

Maxillary brown tumor associated with chronic kidney failure: a case report / Tumor marrom na maxila associado à insuficiência renal crônica: relato de caso

The brown tumor is a bone lesion that may affect the entire skeleton, including the maxillary bones. These tumors are characterized as focal giant cell lesions that may be associated with primary or secondary hyperparathyroidism (HPT). Brown tumors are invasive in some cases and an association with chronic renal failure (CRF) has been reported. With the aim to facilitate the differential diagnosis of bone lesions that may affect dialysis patients, this paper describes a case of brown tumor in a 36- year old patient with CRF, secondary HPT carrier, who had a lesion on the right maxilla for approximately five months.

O tumor marrom é uma lesão óssea que pode acometer todo o esqueleto, inclusive os ossos maxilares. Apresenta-se como uma lesão focal de células gigantes associada ao hiperparatireoidismo (HPT) primário ou secundário; é invasiva em alguns casos e pode associar-se à insuficiência renal crônica (IRC). O presente trabalho descreve um caso de tumor marrom em uma paciente de 36 anos com insuficiência renal crônica (IRC), portadora de HPT secundário, que apresentava lesão, com duração de aproximadamente cinco meses, localizada na maxila direita, com o objetivo de auxiliar o diagnóstico diferencial das lesões ósseas que podem afetar pacientes dialíticos.

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.

Brown Tumor of the Humerus Associated with Secondary Hyperparathyroidism: A Case Report of Successful Treatment after Subtotal Parathyroidectomy / 대한내분비외과학회지

Brown tumor is a bone disease that arises in the setting of increased osteoclastic activity and fibroblastic proliferation in the involved bone. It is well recognized as serious complication of hyperparathyroidism. Brown tumor is uncommon, and brown tumor with secondary hyperparathyroidism resulting from chronic renal failure has rarely been reported. We recently experienced a case of a 28-year-old Korean woman with chronic renal failure caused by chronic glomerulonephritis, on hemodialysis for nine years. She has been hospitalized with left shoulder pain for two years. Image studies showed multiple cystic masses, and both suspicious marked thinning and partial destruction of the cortex on the head of the left humerus. Histopathologic analysis of the mass lesion showed a fibrotic capsule, hemosiderin pigmentation, and giant cell, all characteristic of brown tumor. A subtotal parathyroidectomy was done without surgery of the bony lesion (brown tumor), with successful results. We report this case with a brief review of the literature.

Seven Year-follow-up of a Brown Tumor in the Maxilla Associated with Secondary Hyperparathyroidism in End-stage Renal Failure / 대한골다공증학회지

Hyperparathyroidism is a frequent complication of chronic kidney disease (CKD) as a result of prolonged hyperphosphatemia and hypocalcemia. Brown tumor is a rare bony complication of hyperparathyroidism as a result of increased osteoclastic activity and fibroblastic proliferation. Frequent sites of brown tumor are known as ribs, clavicles, mandible, and pelvic bone, but maxilla is very rare site. Twenty seven-year-old woman with stage V CKD on hemodialysis presented with maxillary mass which had gradually increased in size for 3 years. It was painless, but tooth derangement occurred. Initial laboratory findings revealed hypercalcemia (11.0 mg/dL), hyperphosphatemia (6.9 mg/dL), high creatinine (7.5 mg/dL), and high serum PTH (1729.9 pg/mL). The bone mineral density was significantly low (lumbar spine Z-score:

A Case of Vitamin D Deficiency with Secondary Hyperparathyroidism and Brown Tumor / 대한골다공증학회지

The importance of vitamin D for skeletal health is well known. Vitamin D deficiency causes secondary hyperparathyroidism, high bone turnover, bone loss, and osteoporotic fractures. We report a case of a 60-year-old women presenting with brown tumor in the proximal femur secondary to vitamin D deficiency and secondary hyperparathyroidism. She took calcium supplements and vitamin D3 for 1 year and half dose of risedronate was prescribed for 5 months. After 1 year, pain in the pelvis was much improved and Bone mineral density of the lumbar spine and femoral neck increased by 37% and 29%, respectively.

Hiperparatiroidismo terciario: a propósito de tres pacientes con complicaciones diferentes / Tertiary hyperparathyroidism: representation of the complications in three different cases

El hiperparatiroidismo terciario ha sido reconocido como una hiperfunción autónoma de las glándulas paratiroides. Cuando esta enfermedad se instaura, la única opción terapéutica efectiva es la paratiroidectomía. Estos pacientes frecuentemente tienen hiperplasia paratiroidea por lo que la exploración cervical debe ser bilateral. Presentación de casos: Presentamos 3 pacientes, cada uno con una complicación diferente a propósito del hiperparatiroidismo terciario. La primera paciente con arteriolopatía cálcico-urémica, la segunda con calcinosis tumoral de la cadera, y la tercera paciente con tumor pardo bimaxilar. A todos los pacientes los tratamos con paratiroidectomía total con auto trasplante de la glándula más sana en el músculo recto anterior del abdomen. El estudio anatomopatológico reveló hiperplasia de las glándulas paratiroideas resecadas en todos los pacientes. La complicación principal de este procedimiento fue la hipocalcemia sintomática, requiriendo infusión de calcio endovenoso. Dos pacientes presentaron síndrome de hueso hambriento, que se resolvió progresivamente y en el seguimiento se observó normocalcemia. Hubo desenlace fatal en 2 pacientes como consecuencia de complicaciones sistémicas. El hiperparatiroidismo terciario es una enfermedad rara que el clínico debe reconocer para tratarla oportunamente. La paratiroidectomía total es terapéuticamente efectiva en esta condición, pero la hipocalcemia profunda posoperatoria es frecuente y necesita de un manejo cuidadoso.

Tertiary hyperparathyroidism has been recognized as an autonomous hyperfunction of the parathyroid glands. When this disease is established, the only effective therapeutic option is parathyroidectomy. These patients often have parathyroid hyperplasia so that the the neck exploration must be bilateral. Cases presentation: We report 3 patients, each with a different complication as a consequence of tertiary hyperparathyroidism. The first patient with calcic uremic arteriolopathy, the second with tumoral calcinosis of the hip, and the third patient with bimaxillary brown tumor. We treated all this patients with total parathyroidectomy, followed by healthy gland autotransplantation in the anterior rectus abdominal muscle. The anatomopathological study revealed hyperplasia of the resected parathyroid glands, in all the specimens. The main complication of this procedure were the symptomatic hypocalcemia, requiring intravenous calcium infusion. Two patients had the hungry bone syndrome, which was solved progressively. Two parients died due to systemic complications. Tertiary hyperparathyroidism is a rare disease that the clinician should recognize in order to treat it promptly. Total parathyroidectomy is therapeutically effective in this condition, but the postoperative profound hypocalcemia is frequent and needs a careful management.

A Case of Brown Tumor of the Maxilla Associated with Secondary Hyperparathyroidism / 대한이비인후과학회지

Brown tumors are focal bone lesions caused by increased osteoclastic activity and fibroblastic proliferation encountered in primary, or more rarely, secondary hyperparathyroidism. Ninety-two percent of the patients who undergo dialysis develop secondary hyperparathyroidism. Of these, approximately 1.5% develops brown tumors. We recently experienced a case of brown tumor of the left maxilla that occurred in association with secondary hyperparathyroidism in a 56-year-old female. We present the clinical characteristics, radiological and pathological findings, and treatment of this rare disease entity with a literature review.

Tumor pardo bimaxiliar superior e hiperparatiroidismo terciario: reporte de un caso y revisión de la literatura / Brown tumor bimaxiliar upper and tertiary hyperparathyroidism: a case report and literature review

El tumor pardo u osteoclastoma constituye una manifestación de la osteítis fibrosa quística, siendo esta última el estadio terminal del proceso de remodelación ósea que ocurre en el hiperparatiroidismo. Su localización más frecuente es en los huesos largos, siendo la afectación de los maxilares poco común. Se presenta el caso de una paciente de 37 años de edad, con enfermedad renal crónica e hiperparatiroidismo terciario con tumor pardo bimaxilar. Se discuten los aspectos clínicos radiológicos y terapéuticos de estos procesos.

Brown tumor or osteoclastoma, is a manifestation of osteitis fibrosa cystica, which represents the terminal stage of the bone remodeling process in hyperparathyroidism. Brown tumors are preferentially situated in the long bones, being maxillary presentation very rare. We present a 37 year old female patient with chronic renal disease and tertiary hyperparathyroidism with bimaxillary brown tumor. Clinical, radiological and therapeutic aspects of these processes are discussed.