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Miyaoka D, Imanishi Y, Yano M, et al. Effects of burosumab on osteocalcin and bone mineral density in patient with 15-year history of nonremission tumor-induced osteomalacia initially treated with conventional therapy: Case report. Bone Rep. 2020;13:100736doi: 10.1016/j.bonr.2020.100736.
Miyaoka, D., Imanishi, Y., Yano, M., Toi, N., Nagata, Y., Kurajoh, M., Yamada, S., Morioka, T., & Emoto, M. (2020). Effects of burosumab on osteocalcin and bone mineral density in patient with 15-year history of nonremission tumor-induced osteomalacia initially treated with conventional therapy: Case report. Bone reports, 13100736. https://doi.org/10.1016/j.bonr.2020.100736
Miyaoka, Daichi, et al. "Effects of burosumab on osteocalcin and bone mineral density in patient with 15-year history of nonremission tumor-induced osteomalacia initially treated with conventional therapy: Case report." Bone reports vol. 13 (2020): 100736. doi: https://doi.org/10.1016/j.bonr.2020.100736
Miyaoka D, Imanishi Y, Yano M, Toi N, Nagata Y, Kurajoh M, Yamada S, Morioka T, Emoto M. Effects of burosumab on osteocalcin and bone mineral density in patient with 15-year history of nonremission tumor-induced osteomalacia initially treated with conventional therapy: Case report. Bone Rep. 2020 Nov 21;13:100736. doi: 10.1016/j.bonr.2020.100736. eCollection 2020 Dec. PMID: 33294501; PMCID: PMC7701316.
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Bone Rep. 2020 Nov 21;13:100736. doi: 10.1016/j.bonr.2020.100736. eCollection 2020 Dec.

Effects of burosumab on osteocalcin and bone mineral density in patient with 15-year history of nonremission tumor-induced osteomalacia initially treated with conventional therapy: Case report.

Bone reports

Daichi Miyaoka, Yasuo Imanishi, Masahiro Yano, Norikazu Toi, Yuki Nagata, Masafumi Kurajoh, Shinsuke Yamada, Tomoaki Morioka, Masanori Emoto

Affiliations

  1. Department of Metabolism, Endocrinology and Molecular Medicine, Osaka City University Graduate School of Medicine, Japan.

PMID: 33294501 PMCID: PMC7701316 DOI: 10.1016/j.bonr.2020.100736

Abstract

Excess fibroblast growth factor 23 (FGF23) causes hypophosphatemic osteomalacia, which is associated with impaired bone matrix mineralization. Tumor-induced osteomalacia (TIO) is a rare paraneoplastic syndrome caused by over-secretion of FGF23 from a tumor. Burosumab, a fully human monoclonal antibody with activities against FGF23, was initially approved in Japan before the rest of the world for treatment of FGF23-associated hypophosphatemic osteomalacia by TIO. We report here a patient with a 15-year history of non-remission TIO initially treated with conventional therapy who was then switched to burosumab treatment. Persistent hypophosphatemia and a relative low level of osteocalcin (bone Gla protein, BGP) compared with bone alkaline phosphatase (BAP) level, indicating poor matrix mineralization, developed during long-term conventional therapy. Repeated surgical and stereotactic body radiation treatments did not result in complete resection of the causable tumor, and bone mineral density (BMD) gradually decreased. Ultimately, burosumab treatment was administered and the serum Pi concentration immediately normalized, while both BGP and BMD also showed a good response. This is first known case report of the detailed efficacy of burosumab for nonremission TIO as an alternative to conventional therapy.

© 2020 The Author(s).

Keywords: BGP; Bone mineral density; Burosumab; Hypophosphatemia; Osteocalcin; Tumor-induced osteomalacia

Conflict of interest statement

YI served as a consultant for Kyowa Kirin Co., Ltd. DM, MY, NT, YN, MK, SY, TM, and ME have no conflicts of interest to report.

References

  1. Am J Surg Pathol. 2004 Jan;28(1):1-30 - PubMed
  2. Osteoporos Int. 2020 Jul;31(7):1383-1387 - PubMed
  3. Osteoporos Sarcopenia. 2018 Dec;4(4):119-127 - PubMed
  4. Nat Rev Dis Primers. 2017 Jul 13;3:17044 - PubMed
  5. Clin Chem Lab Med. 2004 Jan;42(1):90-5 - PubMed
  6. Physiol Rev. 1996 Apr;76(2):593-629 - PubMed
  7. Endocr Pract. 2007 Sep;13(5):458-62 - PubMed
  8. J Bone Miner Metab. 2005;23(3):266-9 - PubMed
  9. Joint Bone Spine. 2020 Jan;87(1):81-83 - PubMed
  10. J Bone Miner Res. 2020 Sep 23;: - PubMed
  11. J Bone Miner Res. 1997 Sep;12(9):1502-11 - PubMed
  12. Calcif Tissue Int. 2009 Apr;84(4):313-23 - PubMed
  13. J Bone Miner Res. 2020 Mar;35(3):469-477 - PubMed
  14. Eur J Endocrinol. 2013 Nov 29;170(1):K1-4 - PubMed
  15. J Bone Miner Res. 2004 Mar;19(3):429-35 - PubMed
  16. Endocr Pract. 2005 Mar-Apr;11(2):108-14 - PubMed
  17. Front Endocrinol (Lausanne). 2020 May 28;11:338 - PubMed
  18. J Bone Miner Metab. 2020 May;38(3):405-411 - PubMed
  19. Endocrine. 2020 Sep;69(3):642-649 - PubMed

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