低磷性骨软化症的临床分析
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摘要
目的探讨低磷性骨软化症(HO)的病因及预后。方法回顾性分析11例HO患者的临床资料。结果 11例患者临床表现为不同程度的骨痛、肌无力及活动受限。实验室检查示低血磷、高尿磷;发射型计算机断层成像(ECT)提示骨质疏松。11例患者中,5例通过影像学检查明确肿瘤定位,并行手术切除。病理检查证实1例为假肌源性血管内皮瘤,2例为梭形细胞瘤,1例为成熟性畸胎瘤,1例为神经鞘瘤。术后随访3~6个月,患者血磷恢复正常,骨痛及肌无力均显著改善。另外,3例患者行CT,分别于右膝关节腘窝处、左腰部、L_4椎体左侧附件发现疑似肿瘤占位,3例PET-CT阴性的患者口服磷制剂治疗;随访3~6个月患者血磷未达标,骨痛及肌无力症状较前减轻。结论不明原因的持续性HO患者应考虑肿瘤相关性HO。手术切除肿瘤后,HO预后良好。
Objective To explore the etiology and prognosis of hypophosphatemic osteomalacia( HO).Methods Clinical data of 11 patients with HO were retrospectively analyzed. Results The clinical manifestations of11 cases were presented varying degrees of bone pain,muscle weakness and limited mobility. Laboratory examination showed phosphorus was low,and urinary phosphorus was high; emission computer tomography( ECT) showed osteoporosis. Of 11 patients,5 patients located tumors by imaging examination,and tumors were treated with surgical resection. Pathologically confirmed 1 case as pseudomyogenic hemangioendothelioma,2 cases as spindle cell tumors,1 case as mature teratoma,1 case as schwannoma. Postoperative follow-up for 3-6 months,blood phosphorus returned to normal,bone pain and muscle weakness were significantly improved. In addition,3 patients performed CT,and tumors were suspected occupying in the right knee at the popliteal fossa,left lumbar,left attachment of L4 vertebral,respectively; 3 patients were negative of PET-CT. These patients were given oral phosphate preparations,after follow-up for 3-6 months,blood phosphorus did not return to normal,while the bone pain and myasthenia symptoms reduced than before. Conclusions Cancer-related HO should be considered in unexplained persistence of HO patient. Patients who succeed in surgical excision have good prognosis.
Objective To explore the etiology and prognosis of hypophosphatemic osteomalacia( HO).Methods Clinical data of 11 patients with HO were retrospectively analyzed. Results The clinical manifestations of11 cases were presented varying degrees of bone pain,muscle weakness and limited mobility. Laboratory examination showed phosphorus was low,and urinary phosphorus was high; emission computer tomography( ECT) showed osteoporosis. Of 11 patients,5 patients located tumors by imaging examination,and tumors were treated with surgical resection. Pathologically confirmed 1 case as pseudomyogenic hemangioendothelioma,2 cases as spindle cell tumors,1 case as mature teratoma,1 case as schwannoma. Postoperative follow-up for 3-6 months,blood phosphorus returned to normal,bone pain and muscle weakness were significantly improved. In addition,3 patients performed CT,and tumors were suspected occupying in the right knee at the popliteal fossa,left lumbar,left attachment of L4 vertebral,respectively; 3 patients were negative of PET-CT. These patients were given oral phosphate preparations,after follow-up for 3-6 months,blood phosphorus did not return to normal,while the bone pain and myasthenia symptoms reduced than before. Conclusions Cancer-related HO should be considered in unexplained persistence of HO patient. Patients who succeed in surgical excision have good prognosis.
引文
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[14]Xia WB,Jiang Y,Li M,et al.Levels and dynamic changes of serum fibroblast growth factor 23 in hypophosphatemic rickets/osteomalacia[J].Chin Med J(Engl),2010,123:1158.
[15]Leaf DE,Pereira RC,Bazari H,et al.Oncogenic osteomalacia due to FGF23-expressing colon adenocarcinoma[J].J Clin Endocrinol Metab,2013,98:887.
[16]Wu YW,Ke YH,Zhang ZL.Clinical analysis of 5 misdiagnosed cases of sporadic hypophosphatemic osteomalacia[J].J Osteoporos,2010,16:760.
[17]Yasuda S,Wada S,Kono S,et al.Tumor-induced osteomalacia:benign tumor recurrence after two surgical resections at two different medical institutions[J].Endocr Pract,2013,19:e97.
[18]巴建明,桑艳红,陆菊明,等.12例肿瘤性低磷骨软化症的临床诊治及术后随访[J].中华内分泌代谢杂志,2011,27:19.
[19]Baidya A,Chowdhury S,Mukhopadhyay S,et al.Hypophosphatemic rickets:A case of recurrent pathological fractures[J].Indian J Endocrinol Metab,2012,16:S402.
[20]Wang XL,Mu YM.Emphasis should be placed on the diagnosis and therapy of tumor induced osteomalacia[J].Chin Med J(Engl),2011,124:163.
[2]Harbeck B,Schoecklmann H,Seekamp A,et al.Tumor-induced osteomalacia successful treatment by radio-guided tumor surgery[J].J Clin Rheumatol,2009,15:31.
[3]Prader A,Illig R,Uehlinger E,et al.Rickets following bone tumor[J].Helv Paediatr Acta,1959,14:554.
[4]Mathis DA,Stehel J,Beshay JE,et al.Intracranial phosphaturic mesenchymal tumors report of 2 cases[J].J Neurosurg,2013,118:903.
[5]Chokyu I,Ishibashi K,Goto T,et al.Oncogenic osteomalacia associated with mesenchymal tumor in the middle cranial fossa:a case report[J].J Med Case Rep,2012,6:181.
[6]Jiang Y,Xia WB,Xing XP,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].J Bone Miner Res,2012,27:1967.
[7]Chong WH,Molinolo AA,Chen CC,et al.Tumor-induced osteomalacia[J].Endocr Relat Cancer,2011,18:R53.
[8]张化冰,潘慧,李方,等.奥曲肽显像诊断肿瘤性骨软化一例[J].中华医学杂志,2005,85:2375.
[9]von Falck C,Rodt T,Rosenthal HA,et al.Ga-68-DOTANOC PET/CT for the detection of a mesenchymal tumor causing oncogenic osteomalacia[J].Eur J Nucl Med Mol Imaging,2008,35:1034.
[10]Graham R,Krishnamurthy S,Oliveira A,et al.Frequent expression of fibroblast growth factor-23(FGF23)mRNA in aneurysmal bone cysts and chondromyxoid fibromas[J].J Clin Pathol,2012,65:907.
[11]Nasu T,Kurisu S,Matsuno S,et al.Tumor-induced hypophosphatemic osteomalacia diagnosed by the combinatory procedures of magnetic resonance imaging and venous sampling for FGF23[J].Intern Med,2008,47:957.
[12]杜洪泉,郑燕,章振林.低磷性骨软化症的临诊应对[J].中华内分泌代谢杂志,2014,30:697.
[13]Folpe AL,Fanburg-Smith JC,Billings SD,et al.Most osteomalacia-associated mesenchymal tumors are a single histopathologic entity:an analysis of 32 cases and a comprehensive review of the literature[J].Am J Surg Pathol,2004,28:1.
[14]Xia WB,Jiang Y,Li M,et al.Levels and dynamic changes of serum fibroblast growth factor 23 in hypophosphatemic rickets/osteomalacia[J].Chin Med J(Engl),2010,123:1158.
[15]Leaf DE,Pereira RC,Bazari H,et al.Oncogenic osteomalacia due to FGF23-expressing colon adenocarcinoma[J].J Clin Endocrinol Metab,2013,98:887.
[16]Wu YW,Ke YH,Zhang ZL.Clinical analysis of 5 misdiagnosed cases of sporadic hypophosphatemic osteomalacia[J].J Osteoporos,2010,16:760.
[17]Yasuda S,Wada S,Kono S,et al.Tumor-induced osteomalacia:benign tumor recurrence after two surgical resections at two different medical institutions[J].Endocr Pract,2013,19:e97.
[18]巴建明,桑艳红,陆菊明,等.12例肿瘤性低磷骨软化症的临床诊治及术后随访[J].中华内分泌代谢杂志,2011,27:19.
[19]Baidya A,Chowdhury S,Mukhopadhyay S,et al.Hypophosphatemic rickets:A case of recurrent pathological fractures[J].Indian J Endocrinol Metab,2012,16:S402.
[20]Wang XL,Mu YM.Emphasis should be placed on the diagnosis and therapy of tumor induced osteomalacia[J].Chin Med J(Engl),2011,124:163.