Sinonasal hemangiopericytoma caused hypophosphatemic osteomalacia A case report

被引:8
作者
Li, Jing [1 ]
Huang, Yuanyuan [1 ]
Yang, Fuzhou [2 ]
Zhang, Qi [1 ]
Chen, Decai [1 ]
Wang, Qin [1 ]
机构
[1] Sichuan Univ, Dept Endocrinol & Metab, West China Hosp, 37 Guoxue Lane, Chengdu 610041, Sichuan, Peoples R China
[2] Yaan Peoples Hosp, Dept Nucl Med, Yaan, Sichuan, Peoples R China
关键词
fibroblast growth factor 23; osteomalacia; phosphate; tumor; TUMOR-INDUCED OSTEOMALACIA; ONCOGENIC OSTEOMALACIA; GLOMANGIOPERICYTOMA; LOCALIZATION;
D O I
10.1097/MD.0000000000013849
中图分类号
R5 [内科学];
学科分类号
1002 ; 100201 ;
摘要
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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页数:5
相关论文
共 24 条
[1]  
Agrawal K, 2015, CLIN NUCL MED, V40, pE6, DOI 10.1097/RLU.0000000000000460
[2]   A haemangiopericytoma of the ethmoid sinus causing oncogenic osteomalacia: a case report and review of the literature [J].
Beech, T. J. ;
Rokade, A. ;
Gittoes, N. ;
Johnson, A. P. .
INTERNATIONAL JOURNAL OF ORAL AND MAXILLOFACIAL SURGERY, 2007, 36 (10) :956-958
[3]  
Brandwein-Gensler M, 2012, HEAD NECK PATHOL, V6, P64, DOI 10.1007/s12105-012-0337-8
[4]   Bilateral subtrochanteric fractures in tumour-induced osteomalacia caused by a nasal haemangiopericytoma [J].
Burnand, Henry ;
Samuels, Abigail ;
Hagan, Ian ;
Sawant, Nitin ;
Mutimer, Jonathan .
HIP INTERNATIONAL, 2012, 22 (02) :227-229
[5]   Oncogenic osteomalacia: loss of hypophosphatemia might be the key to avoid misdiagnosis [J].
Chang, Claudia V. ;
Conde, Sandro J. ;
Luvizotto, Renata A. M. ;
Nunes, Vania S. ;
Bonates, Milla C. ;
Felicio, Andre C. ;
Lindsey, Susan C. ;
Moraes, Flavia H. ;
Tagliarini, Jose V. ;
Mazeto, Glaucia M. F. S. ;
Kopp, Peter ;
Nogueira, Celia R. .
ARQUIVOS BRASILEIROS DE ENDOCRINOLOGIA E METABOLOGIA, 2012, 56 (08) :570-573
[6]   Nasal Hemangiopericytoma Causing Oncogenic Osteomalacia [J].
Cho, Sung Il ;
Do, Nam Yong ;
Yu, Seung Woo ;
Choi, Ji Yun .
CLINICAL AND EXPERIMENTAL OTORHINOLARYNGOLOGY, 2012, 5 (03) :173-176
[7]   68Ga-DOTATATE for Tumor Localization in Tumor-Induced Osteomalacia [J].
El-Maouche, Diala ;
Sadowski, Samira M. ;
Papadakis, Georgios Z. ;
Guthrie, Lori ;
Cottle-Delisle, Candice ;
Merkel, Roxanne ;
Millo, Corina ;
Chen, Clara C. ;
Kebebew, Electron ;
Collins, Michael T. .
JOURNAL OF CLINICAL ENDOCRINOLOGY & METABOLISM, 2016, 101 (10) :3575-3581
[8]  
Florenzano Pablo, 2017, Bone Rep, V7, P90, DOI 10.1016/j.bonr.2017.09.002
[9]   Most osteomalacia-associated mesenchymal tumors are a single histopathologic entity - An analysis of 32 cases and a comprehensive review of the literature [J].
Folpe, AL ;
Fanburg-Smith, JC ;
Billings, SD ;
Bisceglia, M ;
Bertoni, F ;
Cho, JY ;
Econs, MJ ;
Inwards, CY ;
de Beur, SMJ ;
Mentzel, T ;
Montgomery, E ;
Michal, M ;
Miettinen, M ;
Mills, SE ;
Reith, JD ;
O'Connell, JX ;
Rosenberg, AE ;
Rubin, BP ;
Sweet, DE ;
Vinh, TN ;
Wold, LE ;
Wehrli, BM ;
White, FKE ;
Zaino, RJ ;
Weiss, SW .
AMERICAN JOURNAL OF SURGICAL PATHOLOGY, 2004, 28 (01) :1-30
[10]   Oncogenic hypophosphatemic osteomalacia associated with a nasal hemangiopericytoma [J].
Fuentealba, C ;
Pinto, D ;
Ballesteros, F ;
Pacheco, D ;
Boettiger, O ;
Soto, N ;
Fernandez, W ;
Gabler, F ;
Gonzales, G ;
Reginato, AJ .
JCR-JOURNAL OF CLINICAL RHEUMATOLOGY, 2003, 9 (06) :373-379