Cargando…

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,...

Descripción completa

Detalles Bibliográficos
Autores principales: 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
Formato: Online Artículo Texto
Lenguaje:English
Publicado: IOS Press 2023
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
_version_ 1785148192922271744
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
work_keys_str_mv AT delimasilvaelisvanessa oraldantrolenereducesmyalgiaandhyperckemiainachildwithryr1relatedexertionalmyalgiarhabdomyolysis
AT doniskarinacarvalho oraldantrolenereducesmyalgiaandhyperckemiainachildwithryr1relatedexertionalmyalgiarhabdomyolysis
AT machadofabianaritacamara oraldantrolenereducesmyalgiaandhyperckemiainachildwithryr1relatedexertionalmyalgiarhabdomyolysis
AT simaomedeirosleonardo oraldantrolenereducesmyalgiaandhyperckemiainachildwithryr1relatedexertionalmyalgiarhabdomyolysis
AT aschoffcarlosalbertodemoura oraldantrolenereducesmyalgiaandhyperckemiainachildwithryr1relatedexertionalmyalgiarhabdomyolysis
AT desouzacarolinafischingermoura oraldantrolenereducesmyalgiaandhyperckemiainachildwithryr1relatedexertionalmyalgiarhabdomyolysis
AT poswarfabianodeoliveira oraldantrolenereducesmyalgiaandhyperckemiainachildwithryr1relatedexertionalmyalgiarhabdomyolysis
AT sautejonasalexmorales oraldantrolenereducesmyalgiaandhyperckemiainachildwithryr1relatedexertionalmyalgiarhabdomyolysis