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Improvement of severe bone pain and activities of daily living in a patient with tumor-induced osteomalacia following the resection of a FGF23-producing- tumor in the oral cavity

Author(s): Kousuke IBA, Koichi SASAKI, Junichi TAKADA, Takeshi MINOWA, Nobuaki ITO, Seiji FUKUMOTO, Tadashi HASEGAWA, Takuro WADA, Toshihiko YAMASHITA

Journal: Journal of Orthopaedics
ISSN 0972-978X

Volume: 6;
Issue: 4;
Date: 2009;
Original page

Keywords: fibroblast growth factor (FGF 23) | tumor-induced osteomalacia | oral soft tumor | bone pain | phosphate

Fibroblast growth factor 23 (FGF23), a protein capable of inhibiting renal tubular epithelial phosphate transport, is thought to be the underlying mechanism in tumor-induced- osteomalacia. In this report, we present a case of tumor-induced- osteomalacia associated with an oral soft tissue tumor that caused overexpression of FGF23.A 35-year-old man suffered from bone pain for 2 years and he presented the difficulty in walking due to severe bone pain and muscle weakness in his lower limbs. Radiography identified old fractures and severe bone atrophy at multiple bones and bone scintigraphy revealed abnormal tracer uptake. Laboratory findings showed that the serum phosphate level was very low (0.8 mg/dl), and FGF23 serum level was elevated (128.7 pg/ml). We diagnosed the patient as FGF 23-producing tumor-induced hypophosphatemic osteomalacia a. About 2 years after the start of our follow-up, a small soft tumor was found behind of the lower lip. After resection of the tumor, the serum phosphate and FGF23 levels rapidly normalized and the severe bone pain markedly improved. Furthermore, the tissue was stained positive using anti-human FGF23 antibody. In this report, we presented the rare case that FGF 23–producing tumor within the oral cavity inducing osteomalacia with severe clinical symptom including bone pain, multiple fragility fractures, muscle weakness and gait disturbance.
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