Westerberg Per-Anton, Olauson Hannes, Toss Göran, Wikström Björn, Morales Ollallo, Linde Torbjörn, Jonsson Kenneth, Ljunggren Osten, Larsson Tobias E
Department of Medical Sciences, Uppsala University Hospital, Uppsala, Sweden.
Endocr Pract. 2008 Apr;14(3):362-7. doi: 10.4158/EP.14.3.362.
To report on a novel strategy for tumor localization in a 62-year-old man with hypophosphatemic tumor-induced osteomalacia (TIO).
Repeated computed tomographic and magnetic resonance imaging scans failed to localize any tumor in a patient with adult-onset hypophosphatemic osteomalacia. Therefore, venous sampling for fibroblast growth factor-23 (FGF23)--a circulating hormone that has been identified as a causative factor for TIO--in major veins was conducted. Serum FGF23 was measured from collected samples by an intact FGF23 enzyme-linked immunosorbent assay.
Venous sampling suggested a local increase in serum FGF23 in the left femoral vein; this finding prompted performance of octreotide scintigraphy restricted to the left leg. A tumor was located at the lateral condyle of the left femur, which was also confirmed by magnetic resonance imaging. Surgical resection of the tumor normalized the serum phosphorus and 1,25-dihydroxyvitamin D3 levels within 5 to 10 days, and FGF23 declined to normal levels within 24 hours. Histologic analysis supported the diagnosis of a soft-tissue giant cell tumor.
Our study case demonstrates the diagnostic complexity and difficulties in localizing a small tumor in a patient with TIO. Venous sampling for FGF23 may be helpful in tumor localization in sporadic cases of hypophosphatemic osteomalacia, especially when noninvasive diagnostic techniques prove insufficient.
报告一种针对一名患有低磷血症性肿瘤诱导骨软化症(TIO)的62岁男性患者进行肿瘤定位的新策略。
一名成年起病的低磷血症性骨软化症患者,多次计算机断层扫描和磁共振成像扫描均未能定位到任何肿瘤。因此,对主要静脉进行了成纤维细胞生长因子23(FGF23)——一种已被确定为TIO致病因素的循环激素——的静脉采样。通过完整FGF23酶联免疫吸附测定法对采集的样本进行血清FGF23测量。
静脉采样提示左股静脉血清FGF23局部升高;这一发现促使对左腿进行了奥曲肽闪烁扫描。在左股骨外侧髁发现了一个肿瘤,磁共振成像也证实了这一点。肿瘤手术切除后,血清磷和1,25 - 二羟维生素D3水平在5至10天内恢复正常,FGF23在24小时内降至正常水平。组织学分析支持软组织巨细胞瘤的诊断。
我们的研究病例展示了TIO患者中定位小肿瘤的诊断复杂性和困难。FGF23静脉采样可能有助于散发性低磷血症性骨软化症病例的肿瘤定位,尤其是在非侵入性诊断技术证明不足时。