Patient Demographics

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Rishi Raj Pikeville Medical Center, Pikeville, Kentucky, USA

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Samaneh Hasanzadeh Bushehr University of Medical Sciences, Bushehr, Iran

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Mitra Dashtizadeh Bushehr University of Medical Sciences, Bushehr, Iran

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Mohammadreza Kalantarhormozi Bushehr University of Medical Sciences, Bushehr, Iran

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Katayoun Vahdat Bushehr University of Medical Sciences, Bushehr, Iran

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Mohammad Hossein Dabbaghmanesh Shiraz University of Medical Sciences, Shiraz, Iran

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Iraj Nabipour Bushehr University of Medical Sciences, Bushehr, Iran

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Mohammdreza Ravanbod Shiraz University of Medical Sciences, Shiraz, Iran

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Majid Assadi Bushehr University of Medical Sciences, Bushehr, Iran

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Basir Hashemi Shiraz University of Medical Sciences, Shiraz, Iran

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Kamyar Asadipooya University of Kentucky, Lexington, Kentucky, USA

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Summary

Oncogenic osteomalacia secondary to glomus tumor is extremely rare. Localization of causative tumors is critical as surgical resection can lead to a complete biochemical and clinical cure. We present a case of oncogenic osteomalacia treated with resection of glomus tumor. A 39-year-old woman with a history of chronic sinusitis presented with chronic body ache and muscle weakness. Biochemical evaluation revealed elevated alkaline phosphatase hypophosphatemia, increased urinary phosphate excretion, low calcitriol, and FGF23 was unsuppressed suggestive of oncogenic osteomalacia. Diagnostic studies showed increase uptake in multiple bones. Localization with MRI of paranasal sinuses revealed a sinonasal mass with concurrent uptake in the same area on the octreotide scan. Surgical resection of the sinonasal mass was consistent with the glomus tumor. The patient improved both clinically and biochemically postoperatively. Along with the case of oncogenic osteomalacia secondary to a glomus tumor, we have also discussed in detail the recent development in the diagnosis and management of oncogenic osteomalacia.

Learning points

  • Tumor-induced osteomalacia is a rare cause of osteomalacia caused by the secretion of FGF23 from mesenchymal tumors.

  • Mesenchymal tumors causing TIO are often difficult to localize and treat.

  • Resection of the tumor can result in complete resolution of biochemical and clinical manifestations in a very short span of time.

  • Glomus tumor can lead to tumor induced osteomalacia and should be surgically treated.

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Maryam Heidarpour Isfahan University of Medical Sciences Isfahan Endocrine and Metabolism Research Center Ringgold Standard Institution, Isfahan, Iran

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Mehdi Karami Isfahan University of Medical Sciences Isfahan Endocrine and Metabolism Research Center Ringgold Standard Institution, Isfahan, Iran

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Pegah Hedayat Isfahan University of Medical Sciences Isfahan Endocrine and Metabolism Research Center Ringgold Standard Institution, Isfahan, Iran

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Ashraf Aminorroaya Isfahan University of Medical Sciences Isfahan Endocrine and Metabolism Research Center Ringgold Standard Institution, Isfahan, Iran

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Summary

Primary hyperparathyroidism revealed by thoracic spine brown tumor and peptic ulcer bleeding is rare. We presented a case of 33-year-old male patient who was admitted with paraplegia. Thoracic spine magnetic resonance imaging (MRI) showed extradural lesion at T4 level. He underwent surgical decompression in T4. According to histopathologic finding and elevated serum parathormone (PTH) and hypercalcemia (total serum calcium 12.1 mg/dL), the diagnosis of brown tumor was down. Ultrasonography of his neck showed a well-defined lesion of 26 × 14 × 6 mm. The day after surgery, he experienced 2 episodes of melena. Bedside upper gastrointestinal endoscopy showed gastric peptic ulcer with visible vessel. Treatment with intragastric local instillation of epinephrine and argon plasma coagulation was done to stop bleeding. After stabilization of the patient, parathyroidectomy was performed. Histologic study showed the parathyroid adenoma without any manifestation of malignancy. At discharge, serum calcium was normal (8.6 mg/dL). On 40th day of discharge, standing and walking status was normal.

Learning points:

  • Thoracic spine involvement is a very rare presentation of primary hyperparathyroidism.

  • The issue of whether primary hyperparathyroidism increases the risk of peptic ulcer disease remains controversial. However, gastrointestinal involvement has been reported in association with classic severe primary hyperparathyroidism.

  • The treatment of brown tumor varies from case to case.

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