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Efficacy and Safety of Denosumab Therapy for Osteogenesis Imperfecta Patients with Osteoporosis—Case Series

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...

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Autores principales: Kobayashi, Tsukasa, Nakamura, Yukio, Suzuki, Takako, Yamaguchi, Tomomi, Takeda, Ryojun, Takagi, Masaki, Hasegawa, Tomonobu, Kosho, Tomoki, Kato, Hiroyuki
Formato: Online Artículo Texto
Lenguaje:English
Publicado: MDPI 2018
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC6306860/
https://www.ncbi.nlm.nih.gov/pubmed/30477250
http://dx.doi.org/10.3390/jcm7120479
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author Kobayashi, Tsukasa
Nakamura, Yukio
Suzuki, Takako
Yamaguchi, Tomomi
Takeda, Ryojun
Takagi, Masaki
Hasegawa, Tomonobu
Kosho, Tomoki
Kato, Hiroyuki
author_facet Kobayashi, Tsukasa
Nakamura, Yukio
Suzuki, Takako
Yamaguchi, Tomomi
Takeda, Ryojun
Takagi, Masaki
Hasegawa, Tomonobu
Kosho, Tomoki
Kato, Hiroyuki
author_sort Kobayashi, Tsukasa
collection PubMed
description 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.
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spelling pubmed-63068602019-01-02 Efficacy and Safety of Denosumab Therapy for Osteogenesis Imperfecta Patients with Osteoporosis—Case Series Kobayashi, Tsukasa Nakamura, Yukio Suzuki, Takako Yamaguchi, Tomomi Takeda, Ryojun Takagi, Masaki Hasegawa, Tomonobu Kosho, Tomoki Kato, Hiroyuki J Clin Med Article 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. MDPI 2018-11-24 /pmc/articles/PMC6306860/ /pubmed/30477250 http://dx.doi.org/10.3390/jcm7120479 Text en © 2018 by the authors. Licensee MDPI, Basel, Switzerland. This article is an open access article distributed under the terms and conditions of the Creative Commons Attribution (CC BY) license (http://creativecommons.org/licenses/by/4.0/).
spellingShingle Article
Kobayashi, Tsukasa
Nakamura, Yukio
Suzuki, Takako
Yamaguchi, Tomomi
Takeda, Ryojun
Takagi, Masaki
Hasegawa, Tomonobu
Kosho, Tomoki
Kato, Hiroyuki
Efficacy and Safety of Denosumab Therapy for Osteogenesis Imperfecta Patients with Osteoporosis—Case Series
title Efficacy and Safety of Denosumab Therapy for Osteogenesis Imperfecta Patients with Osteoporosis—Case Series
title_full Efficacy and Safety of Denosumab Therapy for Osteogenesis Imperfecta Patients with Osteoporosis—Case Series
title_fullStr Efficacy and Safety of Denosumab Therapy for Osteogenesis Imperfecta Patients with Osteoporosis—Case Series
title_full_unstemmed Efficacy and Safety of Denosumab Therapy for Osteogenesis Imperfecta Patients with Osteoporosis—Case Series
title_short Efficacy and Safety of Denosumab Therapy for Osteogenesis Imperfecta Patients with Osteoporosis—Case Series
title_sort efficacy and safety of denosumab therapy for osteogenesis imperfecta patients with osteoporosis—case series
topic Article
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC6306860/
https://www.ncbi.nlm.nih.gov/pubmed/30477250
http://dx.doi.org/10.3390/jcm7120479
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