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      Multiple Brown tumors in a Case of Primary Hyperparathyroidism with Pathological Fracture in Femur

      case-report

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          Abstract

          Introduction:

          Pathological fractures in long bones are commonly caused by simple bone cyst or Osteogenesis imperfecta in children and by metastatic tumors from primary carcinoma, multiple myeloma, osteoporosis, and bone tumors in adults. Hyperparathyroidism causing pathological fractures, though a well-known entity, is seen infrequently in clinical practice. The fractures occur in the expansile fibro-cystic bone lesions called as “Brown tumor.” The reported case describes a patient with classical features of primary hyperparathyroidism (PHPT) having multiple lytic lesions in pelvis and bilateral femur with pathological fracture. It is being reported due to its rarity.

          Case Report:

          A 28–year-old young married lady presented with chronic dull aching pain in both thighs and difficulty in walking for 2 years. Examination revealed tenderness in both thighs, with antalgic gait. X-ray pelvis with both thighs showed multiple lytic lesions of variable size in both femora and pelvis. Blood investigations showed raised levels of serum calcium, with highly raised levels of serum parathyroid hormone (PTH). Contrast-enhanced computerized tomography (CT) scan of neck demonstrated parathyroid adenoma. The patient admitted for prophylactic nailing for right femur, developed a fracture while indoor and was managed by right proximal femoral nailing, followed by parathyroid adenoma excision. Follow-up showed dramatic clinical and radiological improvement with good healing of fracture. Lytic lesions healed gradually and blood parameters returned to normal. The patient remains asymptomatic at 2 years follow-up.

          Conclusion:

          Advanced case of symptomatic PHPT affecting bones is rare and it should be considered as a differential diagnosis in cases of a solitary and or multiple osteolytic lesions. Serum calcium and PTH level estimation at an early stage prevents missing the diagnosis and progression of disease. Early diagnosis and appropriate treatment help in rapid clinical improvement with almost total reversal of bony changes, thus avoiding devastating complications.

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          Most cited references13

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          Metastatic disease in long bones. A proposed scoring system for diagnosing impending pathologic fractures.

          H Mirels (1989)
          A weighted scoring system is proposed to quantify the risk of sustaining a pathologic fracture through a metastatic lesion in a long bone. This system objectively analyzes and combines four roentgenographic and clinical risk factors into a single score. Retrospective analysis of metastatic long bone lesions was completed in 78 lesions that had been irradiated without prophylactic surgical fixation. Clinical data and roentgenograms were scored prior to irradiation by independent observers. The outcome identified 51 lesions that did not fracture during the subsequent six months and 27 lesions that fractured within six months. A mean score of 7 was found in the nonfracture group, whereas the fracture group had a mean score of 10. The percentage risk of a lesion sustaining a pathologic fracture could be predicted for any given score. As the score increased above 7, so did the percentage risk of fracture. It is suggested that all metastatic lesions in long bones be evaluated prior to irradiation. Lesions with scores of 7 or lower can be safely irradiated without risk of fracture, while lesions with scores of 8 or higher require prophylactic internal fixation prior to irradiation.
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            Bone quality, as measured by trabecular bone score, in patients with primary hyperparathyroidism.

            In primary hyperparathyroidism (PHPT), vertebral fractures (VFx) occur regardless of bone mineral density (BMD) and may depend on decreased bone quality. Trabecular bone score (TBS) is a texture measurement acquired during a spinal dual-energy X-ray absorptiometry (DXA). Recently, TBS has been proposed as an index of bone micro-architecture.
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              Osteitis fibrosa cystica—a forgotten radiological feature of primary hyperparathyroidism

              Summary Although bone disease and stone disease are the universally accepted classical manifestations of primary hyperparathyroidism, clinical parathyroid bone disease is rarely seen today in the United States (<5% of patients) and Western Europe. Nevertheless, in a given patient, classical skeletal involvement can be the first sign of primary hyperparathyroidism, but not recognized because it is not usually included, anymore, in the differential diagnosis of this manifestation of skeletal disease. We describe four cases of primary hyperparathyroidism in which the first clinical manifestation of the disease was a pathological fracture that masqueraded as a malignancy. The presence of large osteolytic lesions gave rise to the initial diagnosis of a primary or metastatic cancer. In none of the reported cases was primary hyperparathyroidism with osteitis fibrosa considered as the diagnosis. It would seem to us that this course is best explained by the fact that in many countries such manifestations of primary hyperparathyroidism have become a rarity. In fact, the incidence of osteitis fibrosa among patients with primary hyperparathyroidism in the US is estimated as so rare, that in majority of medical centers routine x-ray examinations of the bones in these patients is not recommended. The X-ray or computed tomography scan findings of osteitis fibrosa cystica include lytic or multilobular cystic changes. Multiple bony lesions representing brown tumors may be misdiagnosed on computed tomography scan as metastatic carcinoma, bone cysts, osteosarcoma, and especially giant-cell tumor. Distinguishing between primary hyperparathyroidism and malignancy is made readily by the concomitant measurement of parathyroid hormone which in primary hyperparathyroidism, again, will be markedly elevated. In the hypercalcemias of malignancy, such elevations of parathyroid hormone are virtually never seen. Conclusion When radiographic evidence of a lytic lesion and hypercalcemia are present, primary hyperparathyroidism should always be considered in the differential diagnosis.
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                Author and article information

                Journal
                J Orthop Case Rep
                J Orthop Case Rep
                Journal of Orthopaedic Case Reports
                Indian Orthopaedic Research Group (India )
                2250-0685
                2321-3817
                September 2020
                : 10
                : 6
                : 49-53
                Affiliations
                [1 ]Department of Orthopedics, Jawaharlal Nehru Medical College and Acharya Vinoba Bhave Rural Hospital, Datta Meghe Institute of Medical Sciences, Wardha, Maharashtra, India
                [2 ]Department of Orthopedics, Mahatma Gandhi Institute of Medical Sciences, Wardha, Maharashtra, India
                Author notes
                Address of Correspondence: Dr. Nareshkumar Satyanarayan Dhaniwala, Department of Orthopedics, Jawaharlal Nehru Medical College and Acharya Vinoba Bhave Rural Hospital, Datta Meghe Institute of Medical Sciences, Sawangi (Meghe), Wardha - 442 107, Maharashtra. India. E-mail: nsdhaniwala@ 123456gmail.com1
                Article
                JOCR-10-49
                10.13107/jocr.2020.v10.i06.1872
                7815671
                95bc434a-2dce-4dcc-b810-78bf31dc648a
                Copyright: © Indian Orthopaedic Research Group

                This is an open-access article distributed under the terms of the Creative Commons Attribution-Noncommercial-Share Alike 3.0 Unported, which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited.

                History
                Categories
                Case Report

                hyperparathyroidism,brown tumor,osteitis fibrosa cystica,femur,pathological fracture

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