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Oral Dantrolene Reduces Myalgia and Hyperckemia in a Child with RYR1-Related Exertional Myalgia/Rhabdomyolysis
RYR1-related exertional myalgia/rhabdomyolysis (ERM) is an underrecognized condition, which can cause limiting muscle symptoms, and may account for more than one-third of undiagnosed rhabdomyolysis cases. Dantrolene has shown promising results in controlling muscle symptoms in individuals with ERM,...
Autores principales: | , , , , , , , |
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Formato: | Online Artículo Texto |
Lenguaje: | English |
Publicado: |
IOS Press
2023
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Materias: | |
Acceso en línea: | https://www.ncbi.nlm.nih.gov/pmc/articles/PMC10657692/ https://www.ncbi.nlm.nih.gov/pubmed/37781817 http://dx.doi.org/10.3233/JND-230007 |
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author | de Lima Silva, Elis Vanessa Donis, Karina Carvalho Machado, Fabiana Rita Camara Simão Medeiros, Leonardo Aschoff, Carlos Alberto de Moura de Souza, Carolina Fischinger Moura Poswar, Fabiano de Oliveira Saute, Jonas Alex Morales |
author_facet | de Lima Silva, Elis Vanessa Donis, Karina Carvalho Machado, Fabiana Rita Camara Simão Medeiros, Leonardo Aschoff, Carlos Alberto de Moura de Souza, Carolina Fischinger Moura Poswar, Fabiano de Oliveira Saute, Jonas Alex Morales |
author_sort | de Lima Silva, Elis Vanessa |
collection | PubMed |
description | RYR1-related exertional myalgia/rhabdomyolysis (ERM) is an underrecognized condition, which can cause limiting muscle symptoms, and may account for more than one-third of undiagnosed rhabdomyolysis cases. Dantrolene has shown promising results in controlling muscle symptoms in individuals with ERM, however, its use in children remains poorly documented. This case report presents the successful treatment of a 5-year-old patient with ERM using oral dantrolene. The patient experienced notable improvements, including a reduction in the frequency and intensity of myalgia episodes, no hospitalizations due to rhabdomyolysis, a substantial decrease in creatine phosphokinase (CPK) levels, and enhanced performance on the 6-minute walk test. The use of dantrolene was well-tolerated, and no significant adverse effects were observed. This report adds to the existing evidence supporting the effectiveness of oral dantrolene in managing ERM, and, to the best of our knowledge, this is the first report of the use of dantrolene in a pediatric patient for controlling anesthesia-independent muscle symptoms. |
format | Online Article Text |
id | pubmed-10657692 |
institution | National Center for Biotechnology Information |
language | English |
publishDate | 2023 |
publisher | IOS Press |
record_format | MEDLINE/PubMed |
spelling | pubmed-106576922023-11-19 Oral Dantrolene Reduces Myalgia and Hyperckemia in a Child with RYR1-Related Exertional Myalgia/Rhabdomyolysis de Lima Silva, Elis Vanessa Donis, Karina Carvalho Machado, Fabiana Rita Camara Simão Medeiros, Leonardo Aschoff, Carlos Alberto de Moura de Souza, Carolina Fischinger Moura Poswar, Fabiano de Oliveira Saute, Jonas Alex Morales J Neuromuscul Dis Case Report RYR1-related exertional myalgia/rhabdomyolysis (ERM) is an underrecognized condition, which can cause limiting muscle symptoms, and may account for more than one-third of undiagnosed rhabdomyolysis cases. Dantrolene has shown promising results in controlling muscle symptoms in individuals with ERM, however, its use in children remains poorly documented. This case report presents the successful treatment of a 5-year-old patient with ERM using oral dantrolene. The patient experienced notable improvements, including a reduction in the frequency and intensity of myalgia episodes, no hospitalizations due to rhabdomyolysis, a substantial decrease in creatine phosphokinase (CPK) levels, and enhanced performance on the 6-minute walk test. The use of dantrolene was well-tolerated, and no significant adverse effects were observed. This report adds to the existing evidence supporting the effectiveness of oral dantrolene in managing ERM, and, to the best of our knowledge, this is the first report of the use of dantrolene in a pediatric patient for controlling anesthesia-independent muscle symptoms. IOS Press 2023-11-07 /pmc/articles/PMC10657692/ /pubmed/37781817 http://dx.doi.org/10.3233/JND-230007 Text en © 2023 – The authors. Published by IOS Press https://creativecommons.org/licenses/by-nc/4.0/This is an open access article distributed under the terms of the Creative Commons Attribution Non-Commercial (CC BY-NC 4.0) License (https://creativecommons.org/licenses/by-nc/4.0/) , which permits unrestricted non-commercial use, distribution, and reproduction in any medium, provided the original work is properly cited. |
spellingShingle | Case Report de Lima Silva, Elis Vanessa Donis, Karina Carvalho Machado, Fabiana Rita Camara Simão Medeiros, Leonardo Aschoff, Carlos Alberto de Moura de Souza, Carolina Fischinger Moura Poswar, Fabiano de Oliveira Saute, Jonas Alex Morales Oral Dantrolene Reduces Myalgia and Hyperckemia in a Child with RYR1-Related Exertional Myalgia/Rhabdomyolysis |
title | Oral Dantrolene Reduces Myalgia and Hyperckemia in a Child with RYR1-Related Exertional Myalgia/Rhabdomyolysis |
title_full | Oral Dantrolene Reduces Myalgia and Hyperckemia in a Child with RYR1-Related Exertional Myalgia/Rhabdomyolysis |
title_fullStr | Oral Dantrolene Reduces Myalgia and Hyperckemia in a Child with RYR1-Related Exertional Myalgia/Rhabdomyolysis |
title_full_unstemmed | Oral Dantrolene Reduces Myalgia and Hyperckemia in a Child with RYR1-Related Exertional Myalgia/Rhabdomyolysis |
title_short | Oral Dantrolene Reduces Myalgia and Hyperckemia in a Child with RYR1-Related Exertional Myalgia/Rhabdomyolysis |
title_sort | oral dantrolene reduces myalgia and hyperckemia in a child with ryr1-related exertional myalgia/rhabdomyolysis |
topic | Case Report |
url | https://www.ncbi.nlm.nih.gov/pmc/articles/PMC10657692/ https://www.ncbi.nlm.nih.gov/pubmed/37781817 http://dx.doi.org/10.3233/JND-230007 |
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