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

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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.
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