{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,5,4]],"date-time":"2026-05-04T01:12:55Z","timestamp":1777857175915,"version":"3.51.4"},"reference-count":12,"publisher":"SAGE Publications","issue":"6","license":[{"start":{"date-parts":[[2023,9,28]],"date-time":"2023-09-28T00:00:00Z","timestamp":1695859200000},"content-version":"unspecified","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by-nc\/4.0\/"}],"content-domain":{"domain":["journals.sagepub.com"],"crossmark-restriction":true},"short-container-title":["Journal of Neuromuscular Diseases"],"published-print":{"date-parts":[[2023,11,7]]},"abstract":"<jats:p>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.<\/jats:p>","DOI":"10.3233\/jnd-230007","type":"journal-article","created":{"date-parts":[[2023,9,29]],"date-time":"2023-09-29T11:05:17Z","timestamp":1695985517000},"page":"1145-1149","update-policy":"https:\/\/doi.org\/10.1177\/sage-journals-update-policy","source":"Crossref","is-referenced-by-count":4,"title":["Oral Dantrolene Reduces Myalgia and Hyperckemia in a Child with\n                    <i>RYR1<\/i>\n                    -Related Exertional Myalgia\/Rhabdomyolysis"],"prefix":"10.1177","volume":"10","author":[{"given":"Elis Vanessa","family":"de Lima Silva","sequence":"first","affiliation":[{"name":"Graduate Program in Genetics and Molecular Biology, Federal University of Rio Grande do Sul, RS, Brazil"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Karina Carvalho","family":"Donis","sequence":"additional","affiliation":[{"name":"Medical Genetics Service \u2013 Hospital de Cl\u00ednicas de Porto Alegre, RS, Brazil"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Fabiana Rita Camara","family":"Machado","sequence":"additional","affiliation":[{"name":"Physiatry Service \u2013 Hospital de Cl\u00ednicas de Porto Alegre, RS, Brazil"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Leonardo","family":"Sim\u00e3o Medeiros","sequence":"additional","affiliation":[{"name":"Graduate Program in Genetics and Molecular Biology, Federal University of Rio Grande do Sul, RS, Brazil"},{"name":"Medical Genetics Service \u2013 Hospital de Cl\u00ednicas de Porto Alegre, RS, Brazil"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Carlos Alberto de Moura","family":"Aschoff","sequence":"additional","affiliation":[{"name":"Medical Genetics Service \u2013 Hospital de Cl\u00ednicas de Porto Alegre, RS, Brazil"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Carolina Fischinger Moura","family":"de Souza","sequence":"additional","affiliation":[{"name":"Medical Genetics Service \u2013 Hospital de Cl\u00ednicas de Porto Alegre, RS, Brazil"},{"name":"Graduate Program in Child and Adolescent Health, Federal University of Rio Grande do Sul, RS, Brazil"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Fabiano de Oliveira","family":"Poswar","sequence":"additional","affiliation":[{"name":"Graduate Program in Genetics and Molecular Biology, Federal University of Rio Grande do Sul, RS, Brazil"},{"name":"Medical Genetics Service \u2013 Hospital de Cl\u00ednicas de Porto Alegre, RS, Brazil"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Jonas Alex Morales","family":"Saute","sequence":"additional","affiliation":[{"name":"Medical Genetics Service \u2013 Hospital de Cl\u00ednicas de Porto Alegre, RS, Brazil"},{"name":"Graduate Program in Medical Sciences, Federal University of Rio Grande do Sul, RS, Brazil"},{"name":"Internal Medicine Department, Federal University of Rio Grande do Sul, RS, Brazil"}],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"179","published-online":{"date-parts":[[2023,9,28]]},"reference":[{"key":"ref001","doi-asserted-by":"publisher","DOI":"10.1016\/j.nmd.2013.03.008"},{"key":"ref002","doi-asserted-by":"publisher","DOI":"10.1016\/j.neurol.2016.07.018"},{"key":"ref003","doi-asserted-by":"publisher","DOI":"10.1111\/ene.12713"},{"key":"ref004","doi-asserted-by":"publisher","DOI":"10.1016\/j.nmd.2022.06.003"},{"key":"ref005","doi-asserted-by":"publisher","DOI":"10.1213\/00000539-198005000-00011"},{"key":"ref006","doi-asserted-by":"publisher","DOI":"10.1097\/ALN.0000000000000732"},{"key":"ref007","doi-asserted-by":"publisher","DOI":"10.1111\/ene.13051"},{"issue":"3","key":"ref008","first-page":"569","volume":"136","author":"Ibarra Moreno CA","year":"2023","journal-title":"Anesth Analg"},{"key":"ref009","unstructured":"Lexicomp Inc Dantrolene: Drug information [Internet], 2021 [cited 2022 Jan 26]. Available from:https:\/\/www.uptodate-2pt.2ptcom-2pt\/-2ptcontents-2pt\/-2ptdantrolene-2pt\u2013.2ptdrug-2pt\u2013.2ptinformation-2pt?2ptsearch=.2ptdantrolene.2pt&source.2pt=panel.2pt_search.2pt_result.2pt&selectedTitle.2pt=.2pt1\u223c35&usage_type=panel.2pt&k2pt_tab.2pt=drug.2pt_general&display_rank=1"},{"issue":"8","key":"ref010","first-page":"1","volume":"9","author":"Klingler W","year":"2014","journal-title":"Orphanet Journal of Rare Diseases"},{"key":"ref011","doi-asserted-by":"publisher","DOI":"10.1038\/s41436-021-01125-w"},{"key":"ref012","doi-asserted-by":"publisher","DOI":"10.1186\/1471-2466-13-49"}],"container-title":["Journal of Neuromuscular Diseases"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/journals.sagepub.com\/doi\/pdf\/10.3233\/JND-230007","content-type":"application\/pdf","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/journals.sagepub.com\/doi\/full-xml\/10.3233\/JND-230007","content-type":"application\/xml","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/journals.sagepub.com\/doi\/pdf\/10.3233\/JND-230007","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2026,4,29]],"date-time":"2026-04-29T23:52:30Z","timestamp":1777506750000},"score":1,"resource":{"primary":{"URL":"https:\/\/journals.sagepub.com\/doi\/10.3233\/JND-230007"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2023,9,28]]},"references-count":12,"journal-issue":{"issue":"6","published-print":{"date-parts":[[2023,11,7]]}},"alternative-id":["10.3233\/JND-230007"],"URL":"https:\/\/doi.org\/10.3233\/jnd-230007","relation":{},"ISSN":["2214-3599","2214-3602"],"issn-type":[{"value":"2214-3599","type":"print"},{"value":"2214-3602","type":"electronic"}],"subject":[],"published":{"date-parts":[[2023,9,28]]}}}