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Efficacy of denosumab in two cases with multiple-system atrophy and osteoporosis

BACKGROUND: Multiple-system atrophy (MSA) is an α-synucleinopathy with a very aggressive course and poor prognosis, which lacks efficient treatment. Thus, MSA represents a serious health and social problem. Progressive stridor and acute laryngeal obstruction likely occur in MSA; however, little is k...

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Autores principales: Uehara, Masashi, Nakamura, Yukio, Takahashi, Jun, Suzuki, Takako, Kato, Hiroyuki
Formato: Online Artículo Texto
Lenguaje:English
Publicado: Dove Medical Press 2018
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC5939903/
https://www.ncbi.nlm.nih.gov/pubmed/29765223
http://dx.doi.org/10.2147/TCRM.S162574
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author Uehara, Masashi
Nakamura, Yukio
Takahashi, Jun
Suzuki, Takako
Kato, Hiroyuki
author_facet Uehara, Masashi
Nakamura, Yukio
Takahashi, Jun
Suzuki, Takako
Kato, Hiroyuki
author_sort Uehara, Masashi
collection PubMed
description BACKGROUND: Multiple-system atrophy (MSA) is an α-synucleinopathy with a very aggressive course and poor prognosis, which lacks efficient treatment. Thus, MSA represents a serious health and social problem. Progressive stridor and acute laryngeal obstruction likely occur in MSA; however, little is known about the bone metabolism or efficacy of bone absorption drugs, such as denosumab, in osteoporosis with MSA. CASE PRESENTATION AND METHODS: Two male patients with osteoporosis in MSA presented to our institution (at 54 and 68 years of age). Denosumab was started after the diagnosis of osteoporosis in MSA, and the therapy was continued for 18–24 months. RESULTS: Lumbar and hip bone mineral density showed a 3.5% and 0.6% increase at 24 months or a 10.3% and 3.2% increase at 18 months, respectively. Bone turnover markers were also improved in the two cases during follow-up. No fractures or other complications were recorded during the observation period. CONCLUSION: This is the first study describing osteoporosis in two patients with MSA being treated by osteoporotic treatment. Based on our findings, it can be concluded that denosumab may be an effective therapy for osteoporosis in MSA.
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spelling pubmed-59399032018-05-14 Efficacy of denosumab in two cases with multiple-system atrophy and osteoporosis Uehara, Masashi Nakamura, Yukio Takahashi, Jun Suzuki, Takako Kato, Hiroyuki Ther Clin Risk Manag Case Series BACKGROUND: Multiple-system atrophy (MSA) is an α-synucleinopathy with a very aggressive course and poor prognosis, which lacks efficient treatment. Thus, MSA represents a serious health and social problem. Progressive stridor and acute laryngeal obstruction likely occur in MSA; however, little is known about the bone metabolism or efficacy of bone absorption drugs, such as denosumab, in osteoporosis with MSA. CASE PRESENTATION AND METHODS: Two male patients with osteoporosis in MSA presented to our institution (at 54 and 68 years of age). Denosumab was started after the diagnosis of osteoporosis in MSA, and the therapy was continued for 18–24 months. RESULTS: Lumbar and hip bone mineral density showed a 3.5% and 0.6% increase at 24 months or a 10.3% and 3.2% increase at 18 months, respectively. Bone turnover markers were also improved in the two cases during follow-up. No fractures or other complications were recorded during the observation period. CONCLUSION: This is the first study describing osteoporosis in two patients with MSA being treated by osteoporotic treatment. Based on our findings, it can be concluded that denosumab may be an effective therapy for osteoporosis in MSA. Dove Medical Press 2018-05-03 /pmc/articles/PMC5939903/ /pubmed/29765223 http://dx.doi.org/10.2147/TCRM.S162574 Text en © 2018 Uehara et al. This work is published and licensed by Dove Medical Press Limited The full terms of this license are available at https://www.dovepress.com/terms.php and incorporate the Creative Commons Attribution – Non Commercial (unported, v3.0) License (http://creativecommons.org/licenses/by-nc/3.0/). By accessing the work you hereby accept the Terms. Non-commercial uses of the work are permitted without any further permission from Dove Medical Press Limited, provided the work is properly attributed.
spellingShingle Case Series
Uehara, Masashi
Nakamura, Yukio
Takahashi, Jun
Suzuki, Takako
Kato, Hiroyuki
Efficacy of denosumab in two cases with multiple-system atrophy and osteoporosis
title Efficacy of denosumab in two cases with multiple-system atrophy and osteoporosis
title_full Efficacy of denosumab in two cases with multiple-system atrophy and osteoporosis
title_fullStr Efficacy of denosumab in two cases with multiple-system atrophy and osteoporosis
title_full_unstemmed Efficacy of denosumab in two cases with multiple-system atrophy and osteoporosis
title_short Efficacy of denosumab in two cases with multiple-system atrophy and osteoporosis
title_sort efficacy of denosumab in two cases with multiple-system atrophy and osteoporosis
topic Case Series
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC5939903/
https://www.ncbi.nlm.nih.gov/pubmed/29765223
http://dx.doi.org/10.2147/TCRM.S162574
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