Efficacy and safety of denosumab therapy for osteogenesis imperfecta patients with osteoporosis—Case series

Tsukasa Kobayashi, Yukio Nakamura, Takako Suzuki, Tomomi Yamaguchi, Ryojun Takeda, Masaki Takagi, Tomonobu Hasegawa, Tomoki Kosho, Hiroyuki Kato

Research output: Contribution to journalArticlepeer-review

18 Citations (Scopus)


Osteogenesis imperfecta (OI) is a connective tissue disorder that is characterized by low bone density leading to recurrent fractures. The efficacy of the anti-resorption drug denosumab for OI with osteoporosis is still largely unknown. We herein describe the clinical outcomes of eight osteoporotic cases of OI to examine the effects and safety of denosumab. This retrospective, consecutive case series included eight patients respectively aged 42, 40, 14, 22, 3, 51, 37, and 9 years. We measured the bone mineral density (BMD) of the lumbar 1–4 spine (L-BMD) and bilateral hips (H-BMD), bone-specific alkaline phosphatase, urinary type I collagen amino-terminal telopeptide, and tartrate-resistant acid phosphatase 5b before and during denosumab therapy. Despite multiple pretreatment fractures in the cohort, no fractures or severe side effects, such as hypocalcemia, were observed during the observational period apart from a fracture in a young pediatric girl. Both L-BMD and H-BMD were increased by denosumab in seven of eight cases. Bone turnover markers were inhibited in most cases by denosumab therapy. Denosumab treatment could generally raise BMD without any adverse effects. The agent therefore represents a good therapeutic option for OI with osteoporosis.

Original languageEnglish
Article number479
JournalJournal of Clinical Medicine
Issue number12
Publication statusPublished - 2018 Dec 1


  • Bone mineral density
  • Denosumab
  • Fractures
  • Osteogenesis imperfecta
  • Osteoporosis

ASJC Scopus subject areas

  • Medicine(all)


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