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Favorable effects of burosumab on tumor-induced osteomalacia caused by an undetectable tumor A case report

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Title: Favorable effects of burosumab on tumor-induced osteomalacia caused by an undetectable tumor A case report
Authors: Oe, Yuki Browse this author
Kameda, Hiraku Browse this author →KAKEN DB
Nomoto, Hiroshi Browse this author
Sakamoto, Keita Browse this author
Soyama, Takeshi Browse this author
Cho, Kyu Yong Browse this author
Nakamura, Akinobu Browse this author
Iwasaki, Koji Browse this author
Abo, Daisuke Browse this author
Kudo, Kohsuke Browse this author
Miyoshi, Hideaki Browse this author
Atsumi, Tatsuya Browse this author →KAKEN DB
Keywords: burosumab
tumor-induced osteomalacia
undetectable tumor
Issue Date: 19-Nov-2021
Publisher: Lippincott Williams & Wilkins (LWW)
Journal Title: Medicine
Volume: 100
Issue: 46
Start Page: e27895
Publisher DOI: 10.1097/MD.0000000000027895
Abstract: Rationale: Tumor-induced osteomalacia (TIO) is curable by tumor resection, but detection of the tumor can be challenging. Overproduction of fibroblast growth factor 23 (FGF23) by the tumor causes hypophosphatemia and consequently induces inappropriate bone turnover. Conventionally oral phosphate supplementation was the only treatment for TIO, but had risks of hypercalciuria and nephrocalcinosis. Burosumab, a human monoclonal anti-FGF23 antibody, was recently post-marketed in Japan against for FGF23-related hypophosphatemia. Herein, we present a case of TIO with undetectable tumor that was successfully treated with burosumab. Patient concerns: A 47-year-old woman was forced to use a wheelchair because of pain in both feet. Diagnosis: Laboratory findings showed hypophosphatemia, elevated bone markers, and high serum FGF23 without renal tubular defects. Imaging studies revealed bone atrophy in the feet, decreased bone density, and multiple pseudofractures in the talar, sacral, and L5 vertebral regions. After excluding drug-induced and hereditary osteomalacia, we diagnosed her as TIO. Interventions: Comprehensive imaging studies and stepwise venous sampling failed to localize the tumor, and we started to administer subcutaneous burosumab. Outcomes: After administration of burosumab, her serum phosphate was normalized without phosphate supplementation within 2 months. Improvement of pseudofractures, relief of pain evaluated by a visual analog scale, and normalization of bone biomarkers were observed. The patient was able to stand by herself after 6 months administration of burosumab. Lessons: This is the first report in clinical practice to demonstrate favorable effects of burosumab, including not only normalization of serum phosphate but also improvements of pseudofractures and subjective pain, in a patient with TIO and undetectable tumor.
Type: article
Appears in Collections:北海道大学病院 (Hokkaido University Hospital) > 雑誌発表論文等 (Peer-reviewed Journal Articles, etc)

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