踝部肿瘤诱发低磷性骨软化症1例
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摘要
肿瘤源性骨软化症(tumor-induced osteomalacia,TIO)是成人低磷性骨软化症(hypophosphatemic osteomalacia,HO)的重要病因。本文报告1例双侧颈部、胸部肋骨、腰背部、髋膝关节疼痛1年,伴明显低磷血症的患者,通过PET-CT检查发现右踝致病肿瘤,手术切除后病理证实为磷酸盐尿性间叶肿瘤,患者术后血磷逐渐升高,术后1周血磷恢复正常,术后2周骨痛、肌无力缓解,术后2个月症状完全消失。提示PET-CT检查在TIO的诊断中有一定的价值。
Tumor-induced osteomalacia( TIO) is an important cause of hypophosphatemic osteomalacia( HO)in adults. One hypophosphatemia patient complained of pain in bilateral neck,chest ribs,lower back,hip and knee for 1 year was investigated. The tumor was found in right ankle by PET-CT examination. Resection was performed and then phosphaturic mesenchymal tumor was pathologically confirmed. The serum phosphorus concentration gradually increased after tumor resection. Serum phosphorus was back to normal in 1 week of surgery. Bone pain and muscle weakness were alleviated in 2 weeks and completely recovered in 2 months of surgery. It suggests that PET-CT examination has a certain diagnostic value in TIO.
Tumor-induced osteomalacia( TIO) is an important cause of hypophosphatemic osteomalacia( HO)in adults. One hypophosphatemia patient complained of pain in bilateral neck,chest ribs,lower back,hip and knee for 1 year was investigated. The tumor was found in right ankle by PET-CT examination. Resection was performed and then phosphaturic mesenchymal tumor was pathologically confirmed. The serum phosphorus concentration gradually increased after tumor resection. Serum phosphorus was back to normal in 1 week of surgery. Bone pain and muscle weakness were alleviated in 2 weeks and completely recovered in 2 months of surgery. It suggests that PET-CT examination has a certain diagnostic value in TIO.
引文
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[7] Ito N,Shimizu Y,Suzuki H,et al. Clinical utility of systemic venous sampling of FGF23 for identifying tumours responsible for tumour-induced osteomalacia[J]. J Intern Med,2010,268:390-394.
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[11] Hesse E,Moessinger E,Rosenthal H,et al. Oncogenic osteomalacia:exact tumor localization by co-registration of positron emission and computed tomography[J]. J Bone Miner Res,2007,22:158-162.
[12] Roarke MC,Nguyen BD. PET/CT localization of phosphaturic mesenchymal neoplasm causing tumor-induced osteomalacia[J]. Clin Nucl Med, 2007, 32:300-301.
[13] von Falck C,Rodt T,Rosenthal H,et al.(68)GaDOTANOC PET/CT for the detection of a mesenchymal tumor causing oncogenic osteomalacia[J]. Eur J Nucl Med Mol Imaging,2008,35:1034.
[14] Khadgawat R,Singh Y,Kansara S,et al. PET/CT localisation of a scapular haemangiopericytoma with tumour-induced osteomalacia[J]. Singapore Med J,2009,50:e55-57.
[15] Jagtap VS,Sarathi V,Lila AR,et al. Tumor-induced osteomalacia:a single center experience[J]. Endocr Pract,2011,17:177-184.
[16] Ho CL. Ga68-DOTA Peptide PET/CT to Detect Occult Mesenchymal Tumor-Inducing Osteomalacia:A Case Series of Three Patients[J]. Nucl Med Mol Imaging,2015,49:231-236.
[17] Yu WJ,He JW,Fu WZ,et al. Reports of 17 Chinese patients with tumor-induced osteomalacia[J]. J Bone Miner Metab,2016,35:298-307.
[18]金今,邱贵兴,陈宾,等.肿瘤相关性低磷抗D骨软化症的外科干预治疗[J].中国骨质疏松杂志,2005,8:346-348.
[19] Geller JL,Khosravi A,Kelly MH,et al. Cinacalcet in the management of tumor-induced osteomalacia[J]. J Bone Miner Res,2007,22:931-937.
[20] Paglia F,Dionisi S,Minisola S. Octreotide for tumorinduced osteomalacia[J]. N Engl J Med,2002,346:1748-1749.
[21] Fukumoto S. Anti-fibroblast growth factor 23 antibody therapy[J]. Curr Opin Nephrol Hypertens,2014,23:346-351.
[22] Hesse E,Rosenthal H,Bastian L. Radiofrequency ablation of a tumor causing oncogenic osteomalacia[J].N Engl J Med,2007,357:422-424.
[23] Wang H,Zhong D,Liu Y,et al. Surgical treatments of tumor-induced osteomalacia lesions in long bones:seventeen cases with more than one year of follow-up[J]. J Bone Joint Surg Am,2015,97:1084-1094.
[2] Woo VL,Landesberg R,Imel EA,et al. Phosphaturic mesenchymal tumor,mixed connective tissue variant,of the mandible:report of a case and review of the literature[J]. Oral Surg Oral Med Oral Pathol Oral Radiol Endod,2009,108:925-932.
[3] Chong WH,Molinolo AA,Chen CC,et al. Tumor-induced osteomalacia[J]. Endocr Relat Cancer,2011,18:R53-77.
[4]李晔,高鹏.距骨肿瘤相关性低磷抗D骨软化症1例报道[J].中国骨与关节外科,2011,4:81-83.
[5] Nakanishi K,Sakai M,Tanaka H,et al. Whole-body MR imaging in detecting phosphaturic mesenchymal tumor(PMT)in tumor-induced hypophosphatemic osteomalacia[J]. Magn Reson Med Sci,2013,12:47-52.
[6] Jan de Beur SM,Streeten EA,Civelek AC,et al. localisation of mesenchymal tumours by somatostatin receptor imaging[J]. Lancet,2002,359:761-763.
[7] Ito N,Shimizu Y,Suzuki H,et al. Clinical utility of systemic venous sampling of FGF23 for identifying tumours responsible for tumour-induced osteomalacia[J]. J Intern Med,2010,268:390-394.
[8] van Boekel G,Ruinemans-Koerts J,Joosten F,et al.Tumor producing fibroblast growth factor 23 localized by two-staged venous sampling[J]. Eur J Endocrinol,2008,158:431-437.
[9] Ogura E,Kageyama K,Fukumoto S,et al. Development of tumor-induced osteomalacia in a subcutaneous tumor,defined by venous blood sampling of fibroblast growth factor-23[J]. Intern Med, 2008, 47:637-641.
[10] Dupond JL,Mahammedi H,Prie D,et al. Oncogenic osteomalacia:diagnostic importance of fibroblast growth factor 23 and F-18 fluorodeoxyglucose PET/CT scan for the diagnosis and follow-up in one case[J].Bone,2005,36:375-378.
[11] Hesse E,Moessinger E,Rosenthal H,et al. Oncogenic osteomalacia:exact tumor localization by co-registration of positron emission and computed tomography[J]. J Bone Miner Res,2007,22:158-162.
[12] Roarke MC,Nguyen BD. PET/CT localization of phosphaturic mesenchymal neoplasm causing tumor-induced osteomalacia[J]. Clin Nucl Med, 2007, 32:300-301.
[13] von Falck C,Rodt T,Rosenthal H,et al.(68)GaDOTANOC PET/CT for the detection of a mesenchymal tumor causing oncogenic osteomalacia[J]. Eur J Nucl Med Mol Imaging,2008,35:1034.
[14] Khadgawat R,Singh Y,Kansara S,et al. PET/CT localisation of a scapular haemangiopericytoma with tumour-induced osteomalacia[J]. Singapore Med J,2009,50:e55-57.
[15] Jagtap VS,Sarathi V,Lila AR,et al. Tumor-induced osteomalacia:a single center experience[J]. Endocr Pract,2011,17:177-184.
[16] Ho CL. Ga68-DOTA Peptide PET/CT to Detect Occult Mesenchymal Tumor-Inducing Osteomalacia:A Case Series of Three Patients[J]. Nucl Med Mol Imaging,2015,49:231-236.
[17] Yu WJ,He JW,Fu WZ,et al. Reports of 17 Chinese patients with tumor-induced osteomalacia[J]. J Bone Miner Metab,2016,35:298-307.
[18]金今,邱贵兴,陈宾,等.肿瘤相关性低磷抗D骨软化症的外科干预治疗[J].中国骨质疏松杂志,2005,8:346-348.
[19] Geller JL,Khosravi A,Kelly MH,et al. Cinacalcet in the management of tumor-induced osteomalacia[J]. J Bone Miner Res,2007,22:931-937.
[20] Paglia F,Dionisi S,Minisola S. Octreotide for tumorinduced osteomalacia[J]. N Engl J Med,2002,346:1748-1749.
[21] Fukumoto S. Anti-fibroblast growth factor 23 antibody therapy[J]. Curr Opin Nephrol Hypertens,2014,23:346-351.
[22] Hesse E,Rosenthal H,Bastian L. Radiofrequency ablation of a tumor causing oncogenic osteomalacia[J].N Engl J Med,2007,357:422-424.
[23] Wang H,Zhong D,Liu Y,et al. Surgical treatments of tumor-induced osteomalacia lesions in long bones:seventeen cases with more than one year of follow-up[J]. J Bone Joint Surg Am,2015,97:1084-1094.