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Sinonasal hemangiopericytoma caused hypophosphatemic osteomalacia: A case report

RATIONALE: Tumor-induced osteomalacia (TIO) is a rare, paraneoplastic syndrome featured with fibroblast growth factor 23 (FGF23) secretion primarily by benign mesenchymal tumors and sometimes by malignancies. TIO diagnosis and treatment is often delayed because TIO usually has nonspecific generalize...

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Autores principales: Li, Jing, Huang, Yuanyuan, Yang, Fuzhou, Zhang, Qi, Chen, Decai, Wang, Qin
Formato: Online Artículo Texto
Lenguaje:English
Publicado: Wolters Kluwer Health 2018
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC6314754/
https://www.ncbi.nlm.nih.gov/pubmed/30593185
http://dx.doi.org/10.1097/MD.0000000000013849
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author Li, Jing
Huang, Yuanyuan
Yang, Fuzhou
Zhang, Qi
Chen, Decai
Wang, Qin
author_facet Li, Jing
Huang, Yuanyuan
Yang, Fuzhou
Zhang, Qi
Chen, Decai
Wang, Qin
author_sort Li, Jing
collection PubMed
description RATIONALE: Tumor-induced osteomalacia (TIO) is a rare, paraneoplastic syndrome featured with fibroblast growth factor 23 (FGF23) secretion primarily by benign mesenchymal tumors and sometimes by malignancies. TIO diagnosis and treatment is often delayed because TIO usually has nonspecific generalized bone pain and weakness, and location of TIO tumor is quite challenging. Very few TIO caused by sinonasal hemangiopericytoma have been reported in the literature. PATIENT CONCERNS: A 40-year-old Chinese woman presented with diffuse bone pain for more than 1 year. Laboratory examination showed hypophosphatemia, hyperphosphaturia, hypocalcemia, an elevated serum alkaline phosphatase (ALP) level and bone-specific ALP level. Imaging studies revealed low bone mineral density (BMD) and multiple pseudofractures at the ribs. F-18 fluorodeoxyglucose positron emission tomography was negative in searching for tumors. Because no tumor was located, the patient was treated with oral phosphate, calcium, and alfacalcidol, and achieved great relief in her symptoms and improvement in BMD. Six years later, the patient had breast cancer surgery and received chemotherapy, and still had hypophosphatemia. During this time, nasopharyngo-fiberscope showed nasal mass in her left nasal cavity. Then she had her nasal polyps removed and surprisingly the serum phosphate became normal. DIAGNOSES AND INTERVENTIONS: The patient had the nasal mass resected, and pathological diagnosis of the nasal mass was sinonasal hemangiopericytoma. Immunohistochemical analysis was positive for FGF23. Thus the final diagnosis was osteomalacia induced by sinonasal hemangiopericytoma. Phosphate supplementation and alfacalcidol were discontinued. OUTCOMES: The patient had normal serum phosphate after 6-month follow-up. LESSONS: By presenting this case, we hope to remind clinicians that in patients with osteomalacia with undetermined reason and intranasal polypoid mass, sinonasal hemangiopericytoma should be suspected.
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spelling pubmed-63147542019-01-24 Sinonasal hemangiopericytoma caused hypophosphatemic osteomalacia: A case report Li, Jing Huang, Yuanyuan Yang, Fuzhou Zhang, Qi Chen, Decai Wang, Qin Medicine (Baltimore) Research Article RATIONALE: Tumor-induced osteomalacia (TIO) is a rare, paraneoplastic syndrome featured with fibroblast growth factor 23 (FGF23) secretion primarily by benign mesenchymal tumors and sometimes by malignancies. TIO diagnosis and treatment is often delayed because TIO usually has nonspecific generalized bone pain and weakness, and location of TIO tumor is quite challenging. Very few TIO caused by sinonasal hemangiopericytoma have been reported in the literature. PATIENT CONCERNS: A 40-year-old Chinese woman presented with diffuse bone pain for more than 1 year. Laboratory examination showed hypophosphatemia, hyperphosphaturia, hypocalcemia, an elevated serum alkaline phosphatase (ALP) level and bone-specific ALP level. Imaging studies revealed low bone mineral density (BMD) and multiple pseudofractures at the ribs. F-18 fluorodeoxyglucose positron emission tomography was negative in searching for tumors. Because no tumor was located, the patient was treated with oral phosphate, calcium, and alfacalcidol, and achieved great relief in her symptoms and improvement in BMD. Six years later, the patient had breast cancer surgery and received chemotherapy, and still had hypophosphatemia. During this time, nasopharyngo-fiberscope showed nasal mass in her left nasal cavity. Then she had her nasal polyps removed and surprisingly the serum phosphate became normal. DIAGNOSES AND INTERVENTIONS: The patient had the nasal mass resected, and pathological diagnosis of the nasal mass was sinonasal hemangiopericytoma. Immunohistochemical analysis was positive for FGF23. Thus the final diagnosis was osteomalacia induced by sinonasal hemangiopericytoma. Phosphate supplementation and alfacalcidol were discontinued. OUTCOMES: The patient had normal serum phosphate after 6-month follow-up. LESSONS: By presenting this case, we hope to remind clinicians that in patients with osteomalacia with undetermined reason and intranasal polypoid mass, sinonasal hemangiopericytoma should be suspected. Wolters Kluwer Health 2018-12-28 /pmc/articles/PMC6314754/ /pubmed/30593185 http://dx.doi.org/10.1097/MD.0000000000013849 Text en Copyright © 2018 the Author(s). Published by Wolters Kluwer Health, Inc. http://creativecommons.org/licenses/by-nc-nd/4.0 This is an open access article distributed under the terms of the Creative Commons Attribution-Non Commercial-No Derivatives License 4.0 (CCBY-NC-ND), where it is permissible to download and share the work provided it is properly cited. The work cannot be changed in any way or used commercially without permission from the journal. http://creativecommons.org/licenses/by-nc-nd/4.0
spellingShingle Research Article
Li, Jing
Huang, Yuanyuan
Yang, Fuzhou
Zhang, Qi
Chen, Decai
Wang, Qin
Sinonasal hemangiopericytoma caused hypophosphatemic osteomalacia: A case report
title Sinonasal hemangiopericytoma caused hypophosphatemic osteomalacia: A case report
title_full Sinonasal hemangiopericytoma caused hypophosphatemic osteomalacia: A case report
title_fullStr Sinonasal hemangiopericytoma caused hypophosphatemic osteomalacia: A case report
title_full_unstemmed Sinonasal hemangiopericytoma caused hypophosphatemic osteomalacia: A case report
title_short Sinonasal hemangiopericytoma caused hypophosphatemic osteomalacia: A case report
title_sort sinonasal hemangiopericytoma caused hypophosphatemic osteomalacia: a case report
topic Research Article
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC6314754/
https://www.ncbi.nlm.nih.gov/pubmed/30593185
http://dx.doi.org/10.1097/MD.0000000000013849
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