Osteolytic Lesions (Brown Tumors) of Primary Hyperparathyroidism: A Report of Two Cases.

Primary hyperparathyroidism is characterized by excessive production of parathyroid hormone. As the condition progresses, bone loss primarily occurs due to resorption. A complication of this condition is the formation of fibrotic and cystic changes in the bone, known as brown tumors. These lesions occur in areas of significant bone resorption, where fibrovascular tissue and giant cells replace bone tissue, often accompanied by hemorrhage and hemosiderin deposits. These brown lesions are rare, with an occurrence rate ranging from 1.5% to 4.5%. We present two cases of middle-aged women who had presentations consistent with hyperparathyroidism and presented with complications such as bone pain and numbness. Both underwent parathyroidectomy to manage the cause and recovered after the surgery. These cases emphasize the importance of recognizing primary hyperparathyroidism as a potential cause of abnormal lesions and highlight the diverse presentations associated with this condition.

The refractory secondary hyperparathyroidism presenting with retro-orbital brown tumor: a case report.

BACKGROUND: Tertiary hyperparathyroidism describes the autonomous and excessive secretion of parathyroid hormone (PTH) by the parathyroid glands after longstanding secondary hyperparathyroidism in chronic kidney disease. Brown tumors are a sign of uncontrolled hyperparathyroidism. In this case, we have reported a refractory and destructive hyperparathyroidism storm. Also, it presented with atypical onset and unexpected adenoma location. CASE PRESENTATION: A 37-year-old man was diagnosed with end-stage kidney disease 22 years ago. He has been undergoing dialysis treatment since that time. Recently, he was admitted to the ophthalmology department due to the unilateral anterior bulging of the right eye and drooping of the eyelid. Magnetic resonance imaging exhibited an extraconal mass lesion located in the right orbital posterior superolateral position. Computerized tomography scans considered expansile bone lesion with peripheral calcification and originating from the sphenoid wing. The bone mass lesion was resected via craniotomy due to the compressive effect. The pathological findings were consistent with brown tumors. Plasma intact PTH level was 4557 pg/mL. The patient informed that he underwent parathyroidectomy and two leg fractures operation in a medical query. Parathyroid scintigraphy determined three distinct foci consistent with adenomas and one of them was in mediastenum. Second parathyroidectomy was recommended to the patient but the patient refused surgery. Despite his medication and dialysis regimen being revised, PTH levels were maintained at higher levels in follow-up. CONCLUSIONS: We presented a hyperparathyroidism case that was resistant to all treatments and exhibited all the severe complications in a long-term dialysis patient. Furthermore, this case has revealed the importance and difficulty of secondary hyperparathyroidism management.

Primary Hyperparathyroidism Mimicking Skeletal Metastasis - A Diagnostic Dilemma.

Introduction: Primary hyperparathyroidism (PHPT) is an intrinsic abnormality of the parathyroid glands in which there is an inappropriate secretion of parathormone (PTH), resulting in skeletal resorption and bone loss. The characteristic bony changes of fibrotic cystic lesions are called Brown's tumors. Clinical dilemmas exist due to the varied clinical presentation of hypercalcemia with multiple lytic lesions mimicking metastatic bone disease. The 99 mTc sestamibi scanning is the imaging modality of choice used for the preoperative localization of parathyroid adenomas. Surgery provides a definitive treatment, and the bony lesions resolve completely over a period of time. Case Report: We present four cases of PHPT where they presented with multiple lytic lesions and were evaluated for metastatic deposits. The diagnosis was confirmed with a biopsy. They were successfully treated by excision of the parathyroid gland. A high index of suspicion will avoid misdiagnosis and inappropriate treatment. Conclusion: PHPT must be considered as a differential diagnosis for multiple osteolytic bone lesions. Diagnosis can be aided by a thorough clinical examination, including an assessment for neck swelling, and laboratory testing of serum calcium levels and PTH levels. Surgical excision of the hyperactive gland serves as the definitive treatment for this condition, with bony lesions regressing gradually over time.

Brown tumors of the jaw: an atypical manifestation of primary hyperparathyroidism / Tumores pardos de mandíbula: una manifestación atípica de hiperparatiroidismo primario

Abstract Brown tumors represent the terminal stage of bone remodeling processes in primary hyperparathyroidism. Currently they are rare, and typically affect long bones, pelvis and ribs. Brown tumors may be not included in the initial differential diagnosis of bone disease, espe cially when they are present in atypical localizations. We reported two cases of oral brown tumors as the initial presentation of primary hyperparathyroidism. In the first case, a 44-year-old woman presented a painful and sessile lesion of 4x3 cm over the central body of the mandible which progressively increased in 4-month. The second case involved a 23-year-old woman who was referred with a 3-month history of a painful and ulcerated mass of 2 cm arising from left maxilla, episodes of gingival hemorrhage and difficulty of breathing. Both cases were solitary tumors with no evidence of palpable cervical lymphadenopathy. Incisional biopsy of oral tumors resul ted in giant cell and primary hyperparathyroidism was confirmed by laboratory tests. After parathyroidectomy, histology confirmed adenoma in both cases. Although this type of clinical presentation has almost disappeared in the recent decades, brown tumors should be consi dered in the differential diagnosis of bone oral masses.

Resumen Los tumores pardos son raros y, por lo general, afectan huesos largos, pelvis y costillas. Pueden no estar incluidos en el diagnóstico diferencial inicial como manifestación de enfermedad esquelética, es pecialmente cuando se presentan en localizaciones atípicas. Comunicamos dos casos de tumores pardos orales como presentación inicial de hiperparatiroidismo primario. En el primer caso, una mujer de 44 años presentó una lesión dolorosa y sésil de 4 × 3 cm sobre el cuerpo central de la mandíbula que aumentó de tamaño progresivamente en 4 meses. El segundo caso corresponde a una mujer de 23 años que acudió por presentar una masa dolorosa y ul cerada de 2 cm en maxilar izquierdo de 3 meses de evolución, episodios de hemorragia gingival y difi cultad para respirar. Todos fueron tumores solitarios sin evidencia de linfadenopatía cervical palpable. La biopsia incisional de los tumores orales resultó en células gigantes, y las pruebas de laboratorio confirmaron el hiperparatiroidismo primario. Tras la paratiroidectomía, la histología confirmó adenoma en ambos casos. Los tumores pardos representan la etapa terminal de los procesos de remodelación ósea en el hiperparatiroidismo primario. Aunque este tipo de presentación clínica casi ha desaparecido en las últimas décadas, los tumores pardos deben ser considerados en el diagnóstico diferencial de las masas óseas orales.

PARATHYROID ADENOMA PRESENTING AS MULTIPLE BROWN TUMORS AND SEVERE ASYMPTOMATIC HYPERCALCEMIA.

Introduction: Primary hyperparathyroidism may have several presentations, varying from an incidental asymptomatic biochemical finding to gastrointestinal, psychiatric, renal and bone manifestations. Brown tumors are rare non-neoplastic lesions because of abnormal bone metabolism. Herein, we describe a patient who presented with lytic bony lesions and severe asymptomatic hypercalcemia due to parathyroid adenoma. Case presentation: A 38-year-old male presented with multiple painful bony lesions over upper and lower limbs. Radiographs of long bones showed multiple lytic lesions with cortical thinning. Investigations revealed hypercalcemia and hyperparathyroidism. A radionuclide scan showed parathyroid adenoma. The patient was treated for hypercalcemia and a parathyroidectomy was performed. Conclusions: In a patient presenting with multiple bony swellings and asymptomatic hypercalcemia, hyperparathyroidism should be suspected. Parathyroid adenoma is a treatable cause of primary hyperparathyroidism.

Tumor pardo en esqueleto axial de dos pacientes con enfermedad renal crónica en diálisis / Brown tumor in the axial skeleton of two patients with chronic kidney disease in dialysis

Resumen Los tumores pardos son una forma localizada de osteítis fibrosa no neoplásica, secundaria a hiperparatiroidismo (primario o secundario). Hacen parte de las alteraciones del metabolismo mineral y óseo de la enfermedad renal crónica (ERC). Se manifiestan como lesiones líticas, expansivas, asociadas a masas de tejidos blandos, que pueden estar localizadas en cualquier parte del esqueleto, con predilección por las costillas, clavículas, pelvis, fémur, huesos faciales y mandíbula. Reportamos dos casos de paciente con ERC en terapia de reemplazo renal (TRR), con hiperparatiroidismo secundario y tumores pardos localizados en mandíbula, arcos costales y cuerpos vertebrales, con manifestaciones radiológicas atípicas. Conclusión: los tumores pardos hacen parte de las alteraciones óseas de los pacientes con ERC. El aspecto benigno en los estudios de imágenes (lesiones expansivas sin destrucción de la cortical) en el contexto de un paciente con hiperparatiroidismo, pueden sugerir el diagnóstico. (Acta Med Colomb 2018; 43: 221-225).

Abstract Brown tumors are a localized form of non-neoplastic osteitis fibrosa, secondary to hyperparathyroidism (primary or secondary). They are part of the alterations of the mineral and bone metabolism of chronic kidney disease (CKD). They manifest as lytic, expansive lesions associated to soft tissue masses that can be located in any part of the skeleton with predilection for the ribs, clavicles, pelvis, femur, facial bones and jaw. Two cases of patients with CKD in renal replacement therapy (RRT), with secondary hyperparathyroidism and brown tumors located in the jaw, costal arches and vertebral bodies, with atypical radiological manifestations are described. Conclusion: brown tumors are part of the bone disorders of patients with CKD. The benign appearance in imaging studies (expansive lesions without destruction of the cortex) in the context of a patient with hyperparathyroidism, may suggest the diagnosis. (Acta Med Colomb 2018; 43: 221-225).

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.