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

Oe, Yuki; Kameda, Hiraku; Nomoto, Hiroshi et al.

Medicine, 2021, 100(46), e27895

Permalink : https://hdl.handle.net/2115/83674

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https://doi.org/10.1097/MD.0000000000027895     18

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アクセス権
open access
URI
https://hdl.handle.net/2115/83674
タイトル
Favorable effects of burosumab on tumor-induced osteomalacia caused by an undetectable tumor A case report
著者
Oe, Yuki ; Kameda, Hiraku ; Nomoto, Hiroshi ; Sakamoto, Keita ; Soyama, Takeshi ; Cho, Kyu Yong ; Nakamura, Akinobu ; Iwasaki, Koji ; Abo, Daisuke ; Kudo, Kohsuke ; Miyoshi, Hideaki ; Atsumi, Tatsuya
著者名
Oe, Yuki
e-Rad_Researcher
20826127
著者名
Kameda, Hiraku
著者名
Nomoto, Hiroshi
著者名
Sakamoto, Keita
著者名
Soyama, Takeshi
著者名
Cho, Kyu Yong
著者名
Nakamura, Akinobu
著者名
Iwasaki, Koji
著者名
Abo, Daisuke
著者名
Kudo, Kohsuke
著者名
Miyoshi, Hideaki
e-Rad_Researcher
20301905
著者名
Atsumi, Tatsuya
言語
英語
キーワード
burosumab
hypophosphatemia
osteocalcin
treatment
tumor-induced osteomalacia
undetectable tumor
発行日
2021-11-19
出版者
Lippincott Williams & Wilkins (LWW)
収録物名
Medicine
巻(号)
100(46)
ページ
e27895
抄録
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.
資源タイプ
学術雑誌論文
出版タイプ
Version of Record
PISSN
0025-7974
関連情報 (isIdenticalTo)
出版社版DOI
https://doi.org/10.1097/MD.0000000000027895