{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2025,7,30]],"date-time":"2025-07-30T11:49:55Z","timestamp":1753876195347,"version":"3.41.2"},"reference-count":14,"publisher":"Oxford University Press (OUP)","issue":"12","license":[{"start":{"date-parts":[[2023,11,27]],"date-time":"2023-11-27T00:00:00Z","timestamp":1701043200000},"content-version":"vor","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0\/"}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":[],"published-print":{"date-parts":[[2023,12,1]]},"abstract":"<jats:title>Abstract<\/jats:title>\n               <jats:sec>\n                  <jats:title>Background<\/jats:title>\n                  <jats:p>Histiocytoid cardiomyopathy is a rare infancy cardiac disorder manifesting as severe cardiac arrhythmias or dilated cardiomyopathy. There is no specific treatment for these arrhythmias. This is the first report of infantile histiocytoid cardiomyopathy whose refractory ventricular arrhythmias were successfully controlled by high-dose carvedilol.<\/jats:p>\n               <\/jats:sec>\n               <jats:sec>\n                  <jats:title>Case summary<\/jats:title>\n                  <jats:p>A 4-month-old girl presented with asystole, and recurrent ventricular tachycardias. From the histological findings and clinical symptoms, she was diagnosed as histiocytoid cardiomyopathy. Sedatives were the most effective therapy for her arrhythmia, but the cardiac sympathetic denervation was not effective enough. Finally, her ventricular arrhythmias were controlled with high-dose carvedilol, and she was discharged on hospitalization Day 393.<\/jats:p>\n               <\/jats:sec>\n               <jats:sec>\n                  <jats:title>Discussion<\/jats:title>\n                  <jats:p>Carvedilol is the only beta blocker that directly acts on the ryanodine receptor (RyR2) and inhibits store-overload-induced Ca2+ release (SOICR) in myocardium at high dosage. The arrhythmias did not disappear with bisoprolol, landiolol, or verapamil, but high-dose carvedilol was effective. This clinical course suggested that the arrhythmias in histiocytoid cardiomyopathy might be related with SOICR. High-dose carvedilol might be a key drug for patients with histiocytoid cardiomyopathy.<\/jats:p>\n               <\/jats:sec>","DOI":"10.1093\/ehjcr\/ytad588","type":"journal-article","created":{"date-parts":[[2023,11,27]],"date-time":"2023-11-27T23:21:35Z","timestamp":1701127295000},"source":"Crossref","is-referenced-by-count":1,"title":["Case report: high-dose carvedilol as a potential key drug for arrhythmias in histiocytoid cardiomyopathy"],"prefix":"10.1093","volume":"7","author":[{"ORCID":"https:\/\/orcid.org\/0000-0003-3657-683X","authenticated-orcid":false,"given":"Rei","family":"Yoshida","sequence":"first","affiliation":[{"name":"Department of Pediatric Cardiology, National Cerebral and Cardiovascular Center Hospital , 6-1 Kishibeshimmachi, Suita , 5648565 Osaka, Japan"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-7758-840X","authenticated-orcid":false,"given":"Heima","family":"Sakaguchi","sequence":"additional","affiliation":[{"name":"Department of Pediatric Cardiology, National Cerebral and Cardiovascular Center Hospital , 6-1 Kishibeshimmachi, Suita , 5648565 Osaka, Japan"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Yoshiaki","family":"Kato","sequence":"additional","affiliation":[{"name":"Department of Pediatric Cardiology, National Cerebral and Cardiovascular Center Hospital , 6-1 Kishibeshimmachi, Suita , 5648565 Osaka, Japan"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Kenichi","family":"Kurosaki","sequence":"additional","affiliation":[{"name":"Department of Pediatric Cardiology, National Cerebral and Cardiovascular Center Hospital , 6-1 Kishibeshimmachi, Suita , 5648565 Osaka, Japan"}],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"286","published-online":{"date-parts":[[2023,11,27]]},"reference":[{"key":"2024030519571288600_ytad588-B1","first-page":"646","article-title":"On arachnocytosis of the myocardium (a contribution to the problem of rhabdomyoma of the heart)","volume":"71","author":"Voth","year":"1962","journal-title":"Frankf Z Pathol"},{"key":"2024030519571288600_ytad588-B2","doi-asserted-by":"crossref","first-page":"1009","DOI":"10.1016\/0002-8703(94)90601-7","article-title":"Infantile histiocytoid cardiomyopathy: three cases and literature review","volume":"128","author":"Malhotra","year":"1994","journal-title":"Am Heart J"},{"key":"2024030519571288600_ytad588-B3","doi-asserted-by":"crossref","first-page":"225","DOI":"10.1002\/clc.20224","article-title":"Histiocytoid cardiomyopathy: a mitochondrial disorder","volume":"31","author":"Finsterer","year":"2008","journal-title":"Clin Cardiol"},{"key":"2024030519571288600_ytad588-B4","doi-asserted-by":"crossref","first-page":"32","DOI":"10.1097\/PAF.0000000000000289","article-title":"Sudden death in a male infant due to histiocytoid cardiomyopathy: an autopsy case and review of the literature","volume":"38","author":"Xie","year":"2017","journal-title":"Am J Forensic Med Pathol"},{"key":"2024030519571288600_ytad588-B5","doi-asserted-by":"crossref","first-page":"174","DOI":"10.1017\/S1047951113002382","article-title":"The perfect storm? Histiocytoid cardiomyopathy and compound CACNA2D1 and RANGRF mutation in a baby","volume":"25","author":"Cataldo","year":"2015","journal-title":"Cardiol Young"},{"key":"2024030519571288600_ytad588-B6","article-title":"Successful catheter ablation for premature ventricular contractions triggering torsade de pointes in a small infant with histiocytoid cardiomyopathy: a case report","volume":"3","author":"Hirano","year":"2019","journal-title":"Eur Heart J Case Rep"},{"key":"2024030519571288600_ytad588-B7","doi-asserted-by":"crossref","first-page":"e007118","DOI":"10.1161\/CIRCEP.118.007118","article-title":"Effective use of percutaneous stellate ganglion blockade in patients with electrical storm","volume":"12","author":"Tian","year":"2019","journal-title":"Circ Arrhythm Electrophysiol"},{"key":"2024030519571288600_ytad588-B8","doi-asserted-by":"crossref","first-page":"1826","DOI":"10.1161\/01.CIR.0000125523.14403.1E","article-title":"Left cardiac sympathetic denervation in the management of high-risk patients affected by the long-QT syndrome","volume":"109","author":"Schwartz","year":"2004","journal-title":"Circulation"},{"key":"2024030519571288600_ytad588-B9","doi-asserted-by":"crossref","first-page":"e12894","DOI":"10.1111\/petr.12894","article-title":"Measures and changes in heart rate variability in pediatric heart transplant recipients","volume":"21","author":"Williams","year":"2017","journal-title":"Pediatr Transplant"},{"key":"2024030519571288600_ytad588-B10","doi-asserted-by":"crossref","first-page":"1003","DOI":"10.1038\/nm.2406","article-title":"Carvedilol and its new analogs suppress arrhythmogenic store overload induced Ca2+ release","volume":"17","author":"Zhou","year":"2011","journal-title":"Nat Med"},{"key":"2024030519571288600_ytad588-B11","doi-asserted-by":"crossref","first-page":"9109503","DOI":"10.1155\/2018\/9109503","article-title":"Antiarrhythmic effects of carvedilol and flecainide in cardiomyocytes derived from catecholaminergic polymorphic ventricular tachycardia patients","volume":"2018","author":"P\u00f6l\u00f6nen","year":"2018","journal-title":"Stem Cells Int"},{"key":"2024030519571288600_ytad588-B12","doi-asserted-by":"crossref","first-page":"1149","DOI":"10.1016\/j.hrthm.2007.05.017","article-title":"Calcium channel blockers and beta-blockers versus beta-blockers alone for preventing exercise-induced arrhythmias in catecholaminergic polymorphic ventricular tachycardia","volume":"4","author":"Rosso","year":"2007","journal-title":"Heart Rhythm"},{"key":"2024030519571288600_ytad588-B13","doi-asserted-by":"crossref","first-page":"7948","DOI":"10.3390\/ijms23147948","article-title":"Spatial and functional crosstalk between the mitochondrial Na+\u2013Ca2+ exchanger NCLX and the sarcoplasmic reticulum Ca2+ pump SERCA in cardiomyocytes","volume":"23","author":"Takeuchi","year":"2022","journal-title":"Int J Mol Sci"},{"key":"2024030519571288600_ytad588-B14","doi-asserted-by":"crossref","first-page":"2766","DOI":"10.1038\/srep02766","article-title":"The mitochondrial Na+\u2013Ca2+ exchanger, NCLX, regulates automaticity of HL-1 cardiomyocytes","volume":"3","author":"Takeuchi","year":"2013","journal-title":"Sci Rep"}],"container-title":["European Heart Journal - Case Reports"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/academic.oup.com\/ehjcr\/advance-article-pdf\/doi\/10.1093\/ehjcr\/ytad588\/53830550\/ytad588.pdf","content-type":"application\/pdf","content-version":"am","intended-application":"syndication"},{"URL":"https:\/\/academic.oup.com\/ehjcr\/article-pdf\/7\/12\/ytad588\/56844293\/ytad588.pdf","content-type":"application\/pdf","content-version":"vor","intended-application":"syndication"},{"URL":"https:\/\/academic.oup.com\/ehjcr\/article-pdf\/7\/12\/ytad588\/56844293\/ytad588.pdf","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2024,3,5]],"date-time":"2024-03-05T19:57:32Z","timestamp":1709668652000},"score":1,"resource":{"primary":{"URL":"https:\/\/academic.oup.com\/ehjcr\/article\/doi\/10.1093\/ehjcr\/ytad588\/7451517"}},"subtitle":[],"editor":[{"given":"Patrick","family":"Badertscher","sequence":"additional","affiliation":[],"role":[{"role":"editor","vocabulary":"crossref"}]},{"given":"Fabien","family":"Barbieri","sequence":"additional","affiliation":[],"role":[{"role":"editor","vocabulary":"crossref"}]},{"given":"Niklas","family":"Schenker","sequence":"additional","affiliation":[],"role":[{"role":"editor","vocabulary":"crossref"}]}],"short-title":[],"issued":{"date-parts":[[2023,11,27]]},"references-count":14,"journal-issue":{"issue":"12","published-print":{"date-parts":[[2023,12,1]]}},"URL":"https:\/\/doi.org\/10.1093\/ehjcr\/ytad588","relation":{},"ISSN":["2514-2119"],"issn-type":[{"type":"electronic","value":"2514-2119"}],"subject":[],"published-other":{"date-parts":[[2023,12]]},"published":{"date-parts":[[2023,11,27]]},"article-number":"ytad588"}}