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      Brown tumor of the iliac crest initially misdiagnosed as a giant cell tumor of the bone

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          Abstract

          Summary

          Brown tumors (BTs) are expansile osteolytic lesions complicating severe primary hyperparathyroidism (PHPT). Clinical, radiological and histological features of BTs share many similarities with other giant cell-containing lesions of the bone, which can make their diagnosis challenging. We report the case of a 32-year-old man in whom an aggressive osteolytic lesion of the iliac crest was initially diagnosed as a giant cell tumor by biopsy. The patient was scheduled for surgical curettage, with a course of neoadjuvant denosumab. Routine biochemical workup prior to denosumab administration incidentally revealed high serum calcium levels. The patient was diagnosed with PHPT and a parathyroid adenoma was identified. In light of these findings, histological slices of the iliac lesion were reviewed and diagnosis of a BT was confirmed. Follow-up CT-scans performed 2 and 7 months after parathyroidectomy showed regression and re-ossification of the bone lesion. The aim of this case report is to underline the importance of distinguishing BTs from other giant cell-containing lesions of the bone and to highlight the relevance of measuring serum calcium as part of the initial evaluation of osteolytic bone lesions. This can have a major impact on patients’ management and can prevent unnecessary invasive surgical interventions.

          Learning points:
          • Although rare, brown tumors should always be considered in the differential diagnosis of osteolytic giant cell-containing bone lesions.

          • Among giant cell-containing lesions of the bone, the main differential diagnoses of brown tumors are giant cell tumors and aneurysmal bone cysts.

          • Clinical, radiological and histological characteristics can be non-discriminating between brown tumors and giant cell tumors. One of the best ways to distinguish these two diagnoses appears to be through biochemical workup.

          • Differentiating brown tumors from giant cell tumors and aneurysmal bone cysts is crucial in order to ensure better patient care and prevent unnecessary morbid surgical interventions.

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          Most cited references9

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          Primary hyperparathyroidism: review and recommendations on evaluation, diagnosis, and management. A Canadian and international consensus

          The purpose of this review is to assess the most recent evidence in the management of primary hyperparathyroidism (PHPT) and provide updated recommendations for its evaluation, diagnosis and treatment. A Medline search of "Hyperparathyroidism. Primary" was conducted and the literature with the highest levels of evidence were reviewed and used to formulate recommendations. PHPT is a common endocrine disorder usually discovered by routine biochemical screening. PHPT is defined as hypercalcemia with increased or inappropriately normal plasma parathyroid hormone (PTH). It is most commonly seen after the age of 50 years, with women predominating by three to fourfold. In countries with routine multichannel screening, PHPT is identified earlier and may be asymptomatic. Where biochemical testing is not routine, PHPT is more likely to present with skeletal complications, or nephrolithiasis. Parathyroidectomy (PTx) is indicated for those with symptomatic disease. For asymptomatic patients, recent guidelines have recommended criteria for surgery, however PTx can also be considered in those who do not meet criteria, and prefer surgery. Non-surgical therapies are available when surgery is not appropriate. This review presents the current state of the art in the diagnosis and management of PHPT and updates the Canadian Position paper on PHPT. An overview of the impact of PHPT on the skeleton and other target organs is presented with international consensus. Differences in the international presentation of this condition are also summarized.
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            Clinical practice. Primary hyperparathyroidism.

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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.

                Author and article information

                Journal
                Endocrinol Diabetes Metab Case Rep
                Endocrinol Diabetes Metab Case Rep
                EDM
                Endocrinology, Diabetes & Metabolism Case Reports
                Bioscientifica Ltd (Bristol )
                2052-0573
                21 April 2020
                2020
                : 2020
                : 20-0029
                Affiliations
                [1 ]Division of Endocrinology, Department of Medicine , Hôpital Maisonneuve-Rosemont, Montréal, Canada
                [2 ]Division of Orthopedic Surgery, Department of Surgery , Hôpital Maisonneuve-Rosemont, Montréal, Canada
                [3 ]Department of Radiology , Hôpital Maisonneuve-Rosemont, Montréal, Canada
                [4 ]Department of Pathology , Hôpital Maisonneuve-Rosemont, Montréal, Canada
                Author notes
                Correspondence should be addressed to S Hamidi; Email: sarah.hamidi@ 123456umontreal.ca
                Article
                EDM200029
                10.1530/EDM-20-0029
                7219131
                1ad1a601-e1cf-4839-a8a1-c41954d91f5d
                © 2020 The authors

                This work is licensed under a Creative Commons Attribution-NonCommercial-NoDerivatives 4.0 International License..

                History
                : 04 March 2020
                : 23 March 2020
                Categories
                Adult
                Male
                Other
                Canada
                Bone
                Parathyroid
                Bone
                PTH
                Brown Tumour
                Hyperparathyroidism (Primary)
                Parathyroid Adenoma
                Bone lesions
                Hypercalcaemia
                Bone biopsy
                Calcium (serum)
                Histopathology
                CT scan
                PTH
                25-hydroxyvitamin-D3
                MRI
                Phosphate (serum)
                X-ray
                Sestamibi scan
                Parathyroidectomy
                Denosumab
                Cholecalciferol
                Calcium carbonate
                Calcitriol
                Pathology
                Surgery
                Radiology/Rheumatology
                Error in Diagnosis/Pitfalls and Caveats
                Error in Diagnosis/Pitfalls and Caveats

                adult,male,other,canada,bone,parathyroid,pth,brown tumour,hyperparathyroidism (primary),parathyroid adenoma,bone lesions,hypercalcaemia,bone biopsy,calcium (serum),histopathology,ct scan,25-hydroxyvitamin-d3,mri,phosphate (serum),x-ray,sestamibi scan,parathyroidectomy,denosumab,cholecalciferol,calcium carbonate,calcitriol,pathology,surgery,radiology/rheumatology,error in diagnosis/pitfalls and caveats,april,2020

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