北京大学学报(医学版) ›› 2019, Vol. 51 ›› Issue (6): 1169-1172. doi: 10.19723/j.issn.1671-167X.2019.06.034

• 病例报告 • 上一篇    下一篇

磷酸盐尿性间叶瘤继发低磷骨软化症1例

魏慧,刘蕊(),王占辉,姚中强   

  1. 北京大学第三医院风湿免疫科,北京 100191
  • 收稿日期:2019-08-26 出版日期:2019-12-18 发布日期:2019-12-19
  • 通讯作者: 刘蕊 E-mail:marryllr@163.com

Hypophosphatemic osteomalacia caused by urinary mesenchymal tumor: A case

Hui WEI,Rui LIU(),Zhan-hui WANG,Zhong-qiang YAO   

  1. Department of Rheumatology, Peking University Third Hospital, Beijing 100191, China
  • Received:2019-08-26 Online:2019-12-18 Published:2019-12-19
  • Contact: Rui LIU E-mail:marryllr@163.com

关键词: 骨软化症, 低磷血症, 间叶瘤

Abstract:

This case report concerns a 34-year-old woman who had been diagnosed with ankylosing spondylitis (AS), fibromyalgia syndrome (FMS), osteoarthritis (OA), lumbar disc herniation and the like in different hospitals during the past 18 months. She had progressive osteoarthrosis, significant muscle weakness, gait abnormalities in weight-bearing areas, however without typical inflammatory low back pain, while the treatment with non-steroidal anti-inflammatory drugs (NSAIDs) was invalid, with normal inflammation index, negative results for rheumatic factor (RF) and human leukocyte antigen (HLA)-B27, and normal erythrocyte sedimentation rate (ESR) and C-reactive protein (CRP). She had hyphosphatemia, normal serum calcium, 1, 25-(OH)2-D3 reduction, elevated alkaline phosphatase (ALP) and normal parathyroid hormone (PTH), however with elevated urinary phosphorus. Finally, the medial thigh nodule was found in the subcutaneous of her inner leg by careful examination and imaging scans including B-ultrasound and PET/CT. The final pathology confirmed that the nodule was phosphate urinary mesenchymal tumors. After the tumor was removed, the patient was treated with anti-osteoporosis and phosphorus supplementation. The symptoms of bone pain and muscle weakness were alleviated, and hypophosphatemia was corrected. It was confirmed that the patient had low-phosphorus osteomalacia due to tumor. Tumor-induced hypophosphatemia osteomalacia (TIO) was a rare paraneoplastic syndrome which was caused by excessive phosphorus excretion induced by the tumor, and was thus categorized as an acquired hypophosphatemic osteomalacia. TIO had an occult onset and was associated with a high rate of misdiagnosis, although TIO has some typical clinical features. Early diagnosis, correctly positioning of the tumor, and surgical resection can achieve good outcomes.

Key words: Osteomalacia, Hypophosphatemia, Mesenchymoma

中图分类号: 

  • R591.44

图1

骶髂关节磁共振成像示双侧骶髂关节面下骨髓水肿,髂关节面不规整"

图2

超声提示脂肪层内边界清晰的低回声实性为主分叶状结节"

图3

PET/CT示右大腿中段皮下代谢活跃结节(绿箭头)"

[1] 唐宏宇, 王海彬, 何伟 , 等. 髋部磷酸盐尿性间叶瘤致全身低磷性骨软化症4例[J]. 中华关节外科杂志: 电子版, 2018,12(5):727-729.
[2] Chong WH, Molinolo AA, Chen CC , et al. Tumor-induced osteomalacia[J]. Endocr Relat Cancer, 2011,18(3):R53-R77.
[3] Bergwitz C, Collins MT, Kamth RS , et al. Case records of the Massachusetts General Hospital. Case 33-2011. A 56-year-old man with hypophosphatemia[J]. N Engl J Med, 2011,365(17):1625-1635.
[4] 许志阳, 张文明 . 踝部肿瘤诱发低磷性骨软化症1例[J]. 中华骨质疏松和骨矿盐疾病杂志, 2018,11(6):584-587.
[5] 李晔, 高鹏 . 距骨肿瘤相关性低磷抗D骨软化症1例报告[J]. 中国骨与关节外科, 2011,4(1):81-83.
[6] 巴建明, 桑艳红, 陆菊明 , 等. 12例肿瘤性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(1):19-23.
doi: 10.3760/cma.j.issn.1000-6699.2011.01.007
[7] 张化冰, 潘慧, 李方 , 等. 奥曲肽显像诊断肿瘤性骨软化症一例[J]. 中华医学杂志, 2005,85(33):2375-2376.
[8] Cliflon-Bligh RJ, Hofman MS, Duncan E , et al. Improving diagnosis of tumor-induced osteomalacia with Gallium-68 DATATATE PET/CT[J]. J Clin Endocrinol Metab, 2013,98(2):687-694.
[9] Agrawal K, Bhadada S, Mittal BR , et al. Comparison of 18F-FDG and 68Ga DOTATATE PET/CT in localization of tumor causing oncogenic osteomalacia [J]. Clin Nucl Med, 2015,40(1):e6-e10.
[10] 魏伟平, 刘海蔚, 全会标 , 等. 肿瘤性低磷骨软化症一例报道并文献复习[J]. 中国全科医学, 2018,21(13):1629-1632.
[11] 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(3):346-348.
[12] Areses-Trapote R, Lopez-Garcia JA, Ubetagoyena-Arrieta M , et al. Hereditary hypophosphatemic rickets with hypercalciuria: case reports[J]. Nefrologia, 2012,32(4):529-534.
[13] Wang H, Zhang 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(13):1084-1094.
[14] Geller JL, Khoseavi A, Kelly MH , et al. Cinacalcet in the ma-nagement of tumor-induced osteomalacia[J]. Bone Miner Res, 2007,22(6):931-937.
[15] Paglia F, Dionisi S, Minisola S . Octreotide for tumor-induced osteomalacia[J]. N Engl J Med, 2002,346(22):1748-1749.
[16] Fukumoto S . Anti-fibroblast growth factor 23 antibody therapy[J]. Curr Opin Nephrol Hypertens, 2014,23(4):346-351.
[17] Hesse E, Rosenthal H, Bastian L . Radiofrequency ablation of a tumor causing oncogenic osteomalacia[J]. N Engl J Med, 2007,357(4):422-424.
[1] 孙雯,李昂,张俊清,袁振芳. 低剂量阿德福韦酯致Fanconi综合征和低磷骨软化症1例及文献回顾[J]. 北京大学学报(医学版), 2020, 52(5): 975-979.
[2] 柳萍, 那加, 张剑波, 张莹. CD117阳性小肠间质瘤中Ki67和p53蛋白的表达及在预后中的意义[J]. 北京大学学报(医学版), 2003, 35(1): 28-32.
[3] 柳萍, 何群, 张莹. 胃肠道间质瘤的临床病理形态和免疫组织化学特点[J]. 北京大学学报(医学版), 2001, 33(5): 441-445.
Viewed
Full text


Abstract

Cited

  Shared   
  Discussed   
[1] 田增民, 陈涛, Nanbert ZHONG, 李志超, 尹丰, 刘爽. 神经干细胞移植治疗遗传性小脑萎缩的临床研究(英文稿)[J]. 北京大学学报(医学版), 2009, 41(4): 456 -458 .
[2] 郭岩, 谢铮. 用一代人时间弥合差距——健康社会决定因素理论及其国际经验[J]. 北京大学学报(医学版), 2009, 41(2): 125 -128 .
[3] 成刚, 钱振华, 胡军. 艾滋病项目自愿咨询检测的技术效率分析[J]. 北京大学学报(医学版), 2009, 41(2): 135 -140 .
[4] 卢恬, 朱晓辉, 柳世庆, 郑杰, 邱晓彦. 白细胞介素2促进宫颈癌细胞系HeLaS3免疫球蛋白G的表达[J]. 北京大学学报(医学版), 2009, 41(2): 158 -161 .
[5] 袁惠燕, 张苑, 范田园. 离子交换型栓塞微球及其载平阳霉素的制备与性质研究[J]. 北京大学学报(医学版), 2009, 41(2): 217 -220 .
[6] 徐莉, 孟焕新, 张立, 陈智滨, 冯向辉, 释栋. 侵袭性牙周炎患者血清中抗牙龈卟啉单胞菌的IgG抗体水平的研究[J]. 北京大学学报(医学版), 2009, 41(1): 52 -55 .
[7] 董稳, 刘瑞昌, 刘克英, 关明, 杨旭东. 氯诺昔康和舒芬太尼用于颌面外科术后自控静脉镇痛的比较[J]. 北京大学学报(医学版), 2009, 41(1): 109 -111 .
[8] 祁琨, 邓芙蓉, 郭新彪. 纳米二氧化钛颗粒对人肺成纤维细胞缝隙连接通讯的影响[J]. 北京大学学报(医学版), 2009, 41(3): 297 -301 .
[9] 李宏亮*, 安卫红*, 赵扬玉, 朱曦. 妊娠合并高脂血症性胰腺炎行血液净化治疗1例[J]. 北京大学学报(医学版), 2009, 41(5): 599 -601 .
[10] 李伟军, 邢晓芳, 曲立科, 孟麟, 寿成超. PRL-3基因C104S位点突变体和CAAX缺失体的构建及表达[J]. 北京大学学报(医学版), 2009, 41(5): 516 -520 .