Tumor-induced osteomalacia (TIO) is a rare paraneoplastic syndrome that results from tumors that secrete fibroblast growth factor 23 (FGF23). This leads to chronic hypophosphatemia. Burosumab, an anti-FGF23 antibody, is an effective treatment when surgery is not possible; however, it complicates FGF23 measurements and postoperative monitoring. We describe the first case report of TIO in which the responsible tumors were discovered during burosumab therapy and successfully resected. Despite tumor removal, the serum FGF23 levels remained elevated. In the postoperative monitoring of patients treated with burosumab, physicians should focus on the serum and urine levels of phosphate rather than FGF23.
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http://dx.doi.org/10.2169/internalmedicine.4958-24 | DOI Listing |
Intern Med
March 2025
Department of Endocrinology and Diabetes, Fukuoka University School of Medicine, Japan.
Tumor-induced osteomalacia (TIO) is a rare paraneoplastic syndrome that results from tumors that secrete fibroblast growth factor 23 (FGF23). This leads to chronic hypophosphatemia. Burosumab, an anti-FGF23 antibody, is an effective treatment when surgery is not possible; however, it complicates FGF23 measurements and postoperative monitoring.
View Article and Find Full Text PDFJBMR Plus
March 2025
Osteoporosis Center, The University of Tokyo Hospital, Tokyo 113-8655, Japan.
Excess fibroblast growth factor 23 (FGF23), a mature osteocyte-derived phosphaturic hormone, causes chronic hypophosphatemic osteomalacia in adults. This rare condition was recently reported in 2 alcoholic patients, with marked improvement upon cessation of alcohol consumption, suggesting a link between alcohol and FGF23-related hypophosphatemia within the highly limited cases. This study aimed to investigate whether the source of excess FGF23 in alcohol-induced FGF23-related hypophosphatemic osteomalacia is the bone or the other organs.
View Article and Find Full Text PDFNatl Med J India
February 2025
Department of Head and Neck Surgical Oncology, Fortis Hospital, Bannerghatta Road, Bengaluru 560076, Karnataka, India.
Tumour-induced osteomalacia (TIO) is a rare paraneoplastic syndrome caused by increased production of fibroblast growth factor 23 (FGF-23). A 45-year-old man presented to us with progressive weakness over 2 years along with recurrent fractures with minimal trauma. His FGF-23 was found to be above the normal range and DOTATATE positron emission tomography (PET) scan showed a well-defined enhancing soft tissue density involving the left posterior ethmoid with extension to the spheno-ethmoidal recess and sphenoid sinus ostium.
View Article and Find Full Text PDFJ Cutan Pathol
February 2025
Department of Laboratory Medicine and Pathology, Mayo Clinic, Rochester, Minnesota, USA.
We report a case of a primary cutaneous spindle cell sarcoma (SCS) with FN1::FGFR1 fusion. The tumor lacked the typical histologic and immunohistochemical features associated with other FN1-rearranged neoplasms, such as phosphaturic mesenchymal tumors (PMT) and calcified chondroid mesenchymal neoplasms (CCMN). Unlike PMTs, which often feature a cartilaginous matrix and are associated with tumor-induced osteomalacia (TIO), the present case lacked these characteristics and did not show FGF23 mRNA expression.
View Article and Find Full Text PDFCalcif Tissue Int
January 2025
Division of Bone Diseases, Department of Medicine, Geneva University Hospitals, Geneva, Switzerland.
Tumor-induced osteomalacia (TIO) is a rare acquired paraneoplastic syndrome caused by a mesenchymal tumor secreting a phosphaturic hormone called FGF23. Patients present with bone pain, fragility fractures and muscle weakness. Biochemical results show hypophosphatemia, raised serum alkaline phosphatase and reduced calcitriol.
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