BMC Musculoskeletal Disorders | |
Surgical treatment of tumor-induced osteomalacia: a retrospective review of 40 cases with extremity tumors | |
Yong Liu1  Peng Gao1  Gui-xing Qiu1  Jin Jin1  Zhi-jian Sun1  | |
[1] Department of Orthopedics, Peking Union Medical College Hospital, Chinese Academy of Medical Sciences and Peking Union Medical College, Dongcheng District Shuaifuyuan No.1, Beijing 100730, China | |
关键词: Tumor resection; Extremity; Surgical treatment; Tumor-induced osteomalacia; | |
Others : 1134449 DOI : 10.1186/s12891-015-0496-3 |
|
received in 2014-12-04, accepted in 2015-02-11, 发布年份 2015 | |
【 摘 要 】
Background
Tumor-induced osteomalacia (TIO) is a rare syndrome typically caused by mesenchymal tumors. It has been shown that complete tumor resection may be curative. However, to our knowledge, there has been no report of a large cohort to exam different surgical approaches. This study was aimed to assess outcomes of different surgical options of patients with tumor-induced osteomalacia at a single institution.
Methods
Patients with extremity tumors treated in our hospital from January, 2004 to July, 2012 were identified. The minimum follow-up period was 12 months. Patient’s demography, tumor location, preoperative preparation, type of surgeries were summarized, and clinical outcomes were recorded. Successful treatment was defined as significant symptom improvement, normal serum phosphorus and significant improvement or normalization of bone mineral density at the last follow-up. Differences between patients with soft tissue tumors and bone tumors were compared.
Results
There were 40 (24 male and 16 female) patients identified, with an average age of 44 years. The tumors were isolated in either soft tissue (25 patients) or bone (12 patients) and combined soft tissue and bone invasion was observed in 3 patients. For the primary surgery, tumor resection and tumor curettage were performed. After initial surgical treatment, six patients then received a second surgery. Four patients were found to have malignant tumors base on histopathology. With a minimum follow-up period of 12 months, 80% of patients (32/40) were treated successfully, including 50% of patients (2/4) with malignant tumors. Compared to patients with bone tumor, surgical results were better in patient with soft tissue tumor.
Conclusions
Surgical treatment was an effective way for TIO. Other than tumor curettage surgery, tumor resection is the preferred options for these tumors.
【 授权许可】
2015 Sun et al.; licensee BioMed Central .
【 预 览 】
Files | Size | Format | View |
---|---|---|---|
20150305212603228.pdf | 579KB | download | |
Figure 1. | 47KB | Image | download |
【 图 表 】
Figure 1.
【 参考文献 】
- [1]Drezner MK: Tumor-induced osteomalacia. Rev Endocr Metab Disord 2001, 2:175-86.
- [2]Chong WH, Molinolo AA, Chen CC, Collins MT: Tumor-induced osteomalacia. Endocr Relat Cancer 2011, 18:R53-77.
- [3]Econs MJ, Drezner MK: Tumor-induced osteomalacia–unveiling a new hormone. N Engl J Med 1994, 330:1679-81.
- [4]Juppner H, Wolf M, Salusky IB: FGF-23: More than a regulator of renal phosphate handling? J Bone Miner Res 2010, 25:2091-7.
- [5]Berndt T, Craig TA, Bowe AE, Vassiliadis J, Reczek D, Finnegan R, et al.: Secreted frizzled-related protein 4 is a potent tumor-derived phosphaturic agent. J Clin Invest 2003, 112:785-94.
- [6]Quarles LD: FGF23, PHEX, and MEPE regulation of phosphate homeostasis and skeletal mineralization. Am J Physiol Endocrinol Metab 2003, 285:E1-9.
- [7]Carpenter TO, Ellis BK, Insogna KL, Philbrick WM, Sterpka J, Shimkets R: Fibroblast growth factor 7: an inhibitor of phosphate transport derived from oncogenic osteomalacia-causing tumors. J Clin Endocrinol Metab 2005, 90:1012-20.
- [8]De Beur SM, Finnegan RB, Vassiliadis J, Cook B, Barberio D, Estes S, et al.: Tumors associated with oncogenic osteomalacia express genes important in bone and mineral metabolism. J Bone Miner Res 2002, 17:1102-10.
- [9]Duet M, Kerkeni S, Sfar R, Bazille C, Liote F, Orcel P: Clinical impact of somatostatin receptor scintigraphy in the management of tumor-induced osteomalacia. Clin Nucl Med 2008, 33:752-6.
- [10]Khadgawat R, Singh Y, Kansara S, Tandon N, Bal C, Seith A, et al.: PET/CT localisation of a scapular haemangiopericytoma with tumour-induced osteomalacia. Singapore Med J 2009, 50:e55-7.
- [11]Andreopoulou P, Dumitrescu CE, Kelly MH, Brillante BA, Cutler PC, Wodajo FM, et al.: Selective venous catheterization for the localization of phosphaturic mesenchymal tumors. J Bone Miner Res 2011, 26:1295-302.
- [12]Ito N, Shimizu Y, Suzuki H, Saito T, Okamoto T, Hori M, et al.: Clinical utility of systemic venous sampling of FGF23 for identifying tumours responsible for tumour-induced osteomalacia. J Intern Med 2010, 268:390-4.
- [13]Jiang Y, Xia WB, Xing XP, Silva BC, Li M, Wang O, et al.: Tumor-induced osteomalacia: an important cause of adult-onset hypophosphatemic osteomalacia in China: Report of 39 cases and review of the literature. J Bone Miner Res 2012, 27:1967-75.
- [14]Khaliq W, Cheripalli P, Tangella K: Tumor-induced osteomalacia (TIO): atypical presentation. South Med J 2011, 104:348-50.
- [15]Folpe AL, Fanburg-Smith JC, Billings SD, Bisceglia M, Bertoni F, Cho JY, et al.: Most osteomalacia-associated mesenchymal tumors are a single histopathologic entity: an analysis of 32 cases and a comprehensive review of the literature. Am J Surg Pathol 2004, 28:1-30.
- [16]Harbeck B, Schocklmann H, Seekamp A, Czech N, Monig H: Tumor-induced osteomalacia: successful treatment by radio-guided tumor surgery. J Clin Rheumatol 2009, 15:31-4.
- [17]Hesse E, Rosenthal H, Bastian L: Radiofrequency ablation of a tumor causing oncogenic osteomalacia. N Engl J Med 2007, 357:422-4.
- [18]Tutton S, Olson E, King D, Shaker JL: Successful Treatment of Tumor-Induced Osteomalacia with CT-Guided Percutaneous Ethanol and Cryoablation. J Clin Endocrinol Metab 2012, 97:3421-5.
- [19]Fuentealba C, Pinto D, Ballesteros F, Pacheco D, Boettiger O, Soto N, et al.: Oncogenic hypophosphatemic osteomalacia associated with a nasal hemangiopericytoma. J Clin Rheumatol 2003, 9:373-9.
- [20]Uramoto N, Furukawa M, Yoshizaki T: Malignant phosphaturic mesenchymal tumor, mixed connective tissue variant of the tongue. Auris Nasus Larynx 2009, 36:104-5.
- [21]Sidell D, Lai C, Bhuta S, Barnes L, Chhetri DK: Malignant phosphaturic mesenchymal tumor of the larynx. Laryngoscope 2011, 121:1860-3.