Yonsei Med J.  2017 Sep;58(5):981-987. 10.3349/ymj.2017.58.5.981.

Localization of Oncogenic Osteomalacia by Systemic Venous Sampling of Fibroblast Growth Factor 23

Affiliations
  • 1Department of Internal Medicine, Endocrine Research Institute, Severance Hospital, Yonsei University College of Medicine, Seoul, Korea. yumie@yuhs.ac
  • 2Imperial College London, London, United Kingdom.
  • 3Department of Radiology, Yonsei University College of Medicine, Seoul, Korea.

Abstract

PURPOSE
Tumor-induced osteomalacia (TIO) is characterized by hypophosphatemia caused by a phosphaturic mesenchymal tumor. While surgical resection of the tumor leads to a cure, identification of the responsible tumor is challenging. Recently, several studies showed that systemic sampling of fibroblast growth factor 23 (FGF23) is helpful for localization of tumors. The present study aimed to evaluate the clinical utility of this method in Korean patients.
MATERIALS AND METHODS
Six patients compatible with TIO who were admitted to our hospital between 2006 and 2015 were analyzed. Systemic venous sampling of FGF23 was performed to detect blind lesions or to confirm a suspicious lesion identified in previous imaging studies.
RESULTS
Venous sampling helped confirming the tumor in five of the six patients. Three patients underwent surgery after sampling, and in two patients, the lesions were detected after 3 years by means of 68Ga-DOTATOC positron emission tomography with computed tomography. In one patient, there was a local elevation of serum FGF23 without any related lesion on additional imaging.
CONCLUSION
Our data strengthened the value of venous sampling of FGF23 in predicting the location of tumors and suggested that it can be more effective in the presence of the relevant lesion in subsequent imaging analyses.

Keyword

Tumor-induced osteomalacia; fibroblast growth factor 23; venous sampling

MeSH Terms

Adolescent
Adult
Fibroblast Growth Factors/*blood
Humans
Magnetic Resonance Imaging
Male
Middle Aged
Neoplasms, Connective Tissue/*blood/diagnostic imaging/*pathology
Octreotide/analogs & derivatives/chemistry
Organometallic Compounds/chemistry
Veins/*pathology
Young Adult
Organometallic Compounds
Fibroblast Growth Factors
Octreotide

Figure

  • Fig. 1 Results of systemic venous sampling of fibroblast growth factor 23 (FGF23) in six patients. (A) Case 1, (B) Case 2, (C) Case 3, (D) Case 4, (E) Case 5, (F) Case 6. (a) Right (Rt.) jugular vein, (b) Left (Lt.) jugular vein, (c) Rt. subclavian vein, (d) Lt. subclavian vein, (e) superior vena cava, (f) inferior vena cava, (g) Rt. common iliac vein, (h) Lt. common iliac vein, (i) Rt. femoral vein, (j) Lt. femoral vein, (k) Rt. popliteal vein, (l) Lt. popliteal vein, (m) Rt. profunda femoris vein, (n) Lt. profunda femoris vein, (o) Rt. peroneal vein, and (p) Rt. anterior tibial vein. Numbers indicate FGF23 concentrations in pg/mL. ‘●’ means the finally confirmed causative tumor.

  • Fig. 2 Imaging data from patients with tumor-induced osteomalacia (TIO). (A) Magnetic resonance imaging (MRI) of the right thigh in case 1 shows a 2.7-cm oval mass within the right gluteus maximus. (B) MRI of the left foot in case 2 reveals a 4.2-cm lobulated mass in the posterior aspect of the ankle. (C) MRI of the left hip in case 3 shows a 0.8-cm, well-defined, round lesion in the left femoral head. (D) F-18 fluorodeoxyglucose positron emission tomography with computed tomography (FDG-PET/CT) in case 3 reveals mild FDG uptake in the left femoral head. (E) 68Ga-DOTATOC PET/CT in case 4 shows increased uptake in the right mandible. (F) MRI of the neck in case 4 with focal enhancement in the right mandible. (G) 68Ga-DOTATOC PET/CT in case 6 shows increased FDG uptake in the left ethmoid and nasal cavity. (H) Computed tomography of the paranasal sinus in case 6 reveals a 1.8-cm soft-tissue lesion in the left ethmoid sinus. Arrows indicate the tumors responsible for TIO.


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