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Abstract
Context
Tumor-induced osteomalacia (TIO) is one of the most common forms of acquired fibroblast growth factor 23 (FGF23)-related hypophosphatemia and is usually caused by phosphaturic mesenchymal tumors (PMTs). Although the complete resection of PMTs can cure TIO, preoperative localization of tumors by standard imaging modalities is often challenging. In addition to 18F-fluoro-2-deoxy-D-glucose positron emission tomography–computed tomography (FDG-PET) and 111In-pentetreotide scintigraphy (SRS), systemic FGF23 venous sampling (FGF23VS) has been used to help localize PMTs in specialized institutions.
Objective
This study aimed to evaluate the diagnostic performance of each imaging test and their combinations in localizing PMTs.
Methods
In an observational retrospective study of patients with adult-onset FGF23-related osteomalacia who underwent all 3 imaging studies (FDG-PET, SRS, and FGF23VS), the rate of successful preoperative localization of the tumors was evaluated only in the patients with pathological diagnoses of PMTs, considering the possibility that pathogenesis of patients without identified tumors might be due to other causes such as late-onset hereditary FGF23-related hypophosphatemia.
Results
A total of 30 Japanese patients with TIO (median age, 60 years [range, 28-87 years]; 10 women [33.3%]) were included in the study. The success rate of preoperative localization for each test and combinations of 2 or 3 tests among 18 patients with PMTs was as follows: 72% (FDG-PET), 72% (SRS), 94% (FGF23VS), 89% (FDG-PET, SRS), 100% (FDG-PET, FGF23VS), 94% (SRS, FGF23VS), and 100% (FDG-PET, SRS, and FGF23VS).
Conclusion
We observed the highest localization rate of PMTs in patients with identified PMTs with the combination of FDG-PET and FGF23VS.
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1 Division of Nephrology and Endocrinology, The University of Tokyo Hospital , Tokyo, 113-8655 , Japan
2 Department of Endocrinology and Diabetes Mellitus, Fukuoka University School of Medicine , Fukuoka, 814-0180 , Japan
3 Division of Molecular Physiology and Metabolism, Faculty of Medicine, Tohoku University , Miyagi, 980-8575 , Japan
4 Department of Orthopedic Surgery, The University of Tokyo Hospital , Tokyo, 113-8655 , Japan
5 Department of Radiology, Graduate School of Medicine, The University of Tokyo , Tokyo, 113-8655 , Japan
6 Department of Diagnostic Imaging and Nuclear Medicine, Graduate School of Medicine Kyoto University , Kyoto, 606-8507 , Japan
7 Department of Pathology, Graduate School of Medicine, The University of Tokyo , Tokyo, 113-8655 , Japan
8 Department of Oral-maxillofacial Surgery, Dentistry and Orthodontics, and Division of Tissue Engineering, The University of Tokyo Hospital , Tokyo, 113-8655 , Japan
9 Fujii Memorial Institute of Medical Sciences, Institute of Advanced Medical Sciences, Tokushima University , Tokushima, 770-8503 , Japan