Carol A Wittlieb-Weber, Brian F Birnbaum, Chesney D Castleberry, Tyler W Cunningham, Paul Esteso, Katheryn E Gambetta, Emily A Hayes, Daphne T Hsu, Beth D Kaufman, Benjamin Kroslowitz, Ashwin K Lal, Angela Lorts, Hugo Martinez, Deepa Mokshagundam, Deipanjan Nandi, John J Parent, Frank Raucci, Nelia Soares, Jonathan H Soslow, Renata Shih, Svetlana Shugh, Chet R Villa, Sarah J Wilkens, Bethany L Wisotzkey, Jennifer Conway
{"title":"心脏药物在治疗杜氏肌营养不良性心肌病中的应用。","authors":"Carol A Wittlieb-Weber, Brian F Birnbaum, Chesney D Castleberry, Tyler W Cunningham, Paul Esteso, Katheryn E Gambetta, Emily A Hayes, Daphne T Hsu, Beth D Kaufman, Benjamin Kroslowitz, Ashwin K Lal, Angela Lorts, Hugo Martinez, Deepa Mokshagundam, Deipanjan Nandi, John J Parent, Frank Raucci, Nelia Soares, Jonathan H Soslow, Renata Shih, Svetlana Shugh, Chet R Villa, Sarah J Wilkens, Bethany L Wisotzkey, Jennifer Conway","doi":"10.1007/s00246-025-03917-2","DOIUrl":null,"url":null,"abstract":"<p><p>This study seeks to understand cardiac medication use in a large cohort of males with Duchenne Muscular Dystrophy (DMD) followed prospectively with focus on current practices and adherence to consensus directed medical therapy (CDMT). DMD patients have been enrolled in the Advanced Cardiac Therapies Improving Outcomes Network (ACTION) Dystrophinopathy Registry since 2021. Cardiac medication use was analyzed at enrollment and most recent follow-up. CDMT was defined as concurrent use of angiotensin-converting-enzyme inhibitor (ACEi)/angiotensin II receptor blocker (ARB) /angiotensin receptor-neprilysin inhibitor (ARNI) plus beta-blocker (BB) plus mineralocorticoid receptor antagonist (MRA). Two hundred sixty-five males with DMD (median age 17.5 (IQR 14.5-21.5) years) were prospectively followed; median follow-up was 11.5 (IQR 6.2-15.6) months. At most recent follow-up, 153 patients (57.7%) had decreased LV systolic function, 67 (25.3% of the cohort) had moderate or severe dysfunction. For patients with moderate or severe dysfunction, CDMT was used for 49/67 (73.1%) at most recent follow-up, similar to 36/51 (70.6%) at enrollment (p = 0.92). Target doses of CDMT were achieved for 27% of males on ACEi/ARB/ARNI, 28% on BB, and 23% on MRA. Initial analysis of a prospective registry of males with DMD showed that ~ 30% of patients with moderate or severe LV dysfunction were not on CDMT at most recent follow-up and the majority did not reach target dosing. Further understanding regarding the optimal combination of cardiac medications for DMD cardiomyopathy is needed, as is a better understanding of the barriers to CDMT optimization given increasing cardiac causes of death for DMD patients.</p>","PeriodicalId":19814,"journal":{"name":"Pediatric Cardiology","volume":" ","pages":""},"PeriodicalIF":1.5000,"publicationDate":"2025-06-20","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":"0","resultStr":"{\"title\":\"Cardiac Medication Use in ACTION for Duchenne Muscular Dystrophy Cardiomyopathy.\",\"authors\":\"Carol A Wittlieb-Weber, Brian F Birnbaum, Chesney D Castleberry, Tyler W Cunningham, Paul Esteso, Katheryn E Gambetta, Emily A Hayes, Daphne T Hsu, Beth D Kaufman, Benjamin Kroslowitz, Ashwin K Lal, Angela Lorts, Hugo Martinez, Deepa Mokshagundam, Deipanjan Nandi, John J Parent, Frank Raucci, Nelia Soares, Jonathan H Soslow, Renata Shih, Svetlana Shugh, Chet R Villa, Sarah J Wilkens, Bethany L Wisotzkey, Jennifer Conway\",\"doi\":\"10.1007/s00246-025-03917-2\",\"DOIUrl\":null,\"url\":null,\"abstract\":\"<p><p>This study seeks to understand cardiac medication use in a large cohort of males with Duchenne Muscular Dystrophy (DMD) followed prospectively with focus on current practices and adherence to consensus directed medical therapy (CDMT). DMD patients have been enrolled in the Advanced Cardiac Therapies Improving Outcomes Network (ACTION) Dystrophinopathy Registry since 2021. Cardiac medication use was analyzed at enrollment and most recent follow-up. CDMT was defined as concurrent use of angiotensin-converting-enzyme inhibitor (ACEi)/angiotensin II receptor blocker (ARB) /angiotensin receptor-neprilysin inhibitor (ARNI) plus beta-blocker (BB) plus mineralocorticoid receptor antagonist (MRA). Two hundred sixty-five males with DMD (median age 17.5 (IQR 14.5-21.5) years) were prospectively followed; median follow-up was 11.5 (IQR 6.2-15.6) months. At most recent follow-up, 153 patients (57.7%) had decreased LV systolic function, 67 (25.3% of the cohort) had moderate or severe dysfunction. For patients with moderate or severe dysfunction, CDMT was used for 49/67 (73.1%) at most recent follow-up, similar to 36/51 (70.6%) at enrollment (p = 0.92). Target doses of CDMT were achieved for 27% of males on ACEi/ARB/ARNI, 28% on BB, and 23% on MRA. Initial analysis of a prospective registry of males with DMD showed that ~ 30% of patients with moderate or severe LV dysfunction were not on CDMT at most recent follow-up and the majority did not reach target dosing. Further understanding regarding the optimal combination of cardiac medications for DMD cardiomyopathy is needed, as is a better understanding of the barriers to CDMT optimization given increasing cardiac causes of death for DMD patients.</p>\",\"PeriodicalId\":19814,\"journal\":{\"name\":\"Pediatric Cardiology\",\"volume\":\" \",\"pages\":\"\"},\"PeriodicalIF\":1.5000,\"publicationDate\":\"2025-06-20\",\"publicationTypes\":\"Journal Article\",\"fieldsOfStudy\":null,\"isOpenAccess\":false,\"openAccessPdf\":\"\",\"citationCount\":\"0\",\"resultStr\":null,\"platform\":\"Semanticscholar\",\"paperid\":null,\"PeriodicalName\":\"Pediatric Cardiology\",\"FirstCategoryId\":\"3\",\"ListUrlMain\":\"https://doi.org/10.1007/s00246-025-03917-2\",\"RegionNum\":4,\"RegionCategory\":\"医学\",\"ArticlePicture\":[],\"TitleCN\":null,\"AbstractTextCN\":null,\"PMCID\":null,\"EPubDate\":\"\",\"PubModel\":\"\",\"JCR\":\"Q3\",\"JCRName\":\"CARDIAC & CARDIOVASCULAR SYSTEMS\",\"Score\":null,\"Total\":0}","platform":"Semanticscholar","paperid":null,"PeriodicalName":"Pediatric Cardiology","FirstCategoryId":"3","ListUrlMain":"https://doi.org/10.1007/s00246-025-03917-2","RegionNum":4,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":null,"EPubDate":"","PubModel":"","JCR":"Q3","JCRName":"CARDIAC & CARDIOVASCULAR SYSTEMS","Score":null,"Total":0}
Cardiac Medication Use in ACTION for Duchenne Muscular Dystrophy Cardiomyopathy.
This study seeks to understand cardiac medication use in a large cohort of males with Duchenne Muscular Dystrophy (DMD) followed prospectively with focus on current practices and adherence to consensus directed medical therapy (CDMT). DMD patients have been enrolled in the Advanced Cardiac Therapies Improving Outcomes Network (ACTION) Dystrophinopathy Registry since 2021. Cardiac medication use was analyzed at enrollment and most recent follow-up. CDMT was defined as concurrent use of angiotensin-converting-enzyme inhibitor (ACEi)/angiotensin II receptor blocker (ARB) /angiotensin receptor-neprilysin inhibitor (ARNI) plus beta-blocker (BB) plus mineralocorticoid receptor antagonist (MRA). Two hundred sixty-five males with DMD (median age 17.5 (IQR 14.5-21.5) years) were prospectively followed; median follow-up was 11.5 (IQR 6.2-15.6) months. At most recent follow-up, 153 patients (57.7%) had decreased LV systolic function, 67 (25.3% of the cohort) had moderate or severe dysfunction. For patients with moderate or severe dysfunction, CDMT was used for 49/67 (73.1%) at most recent follow-up, similar to 36/51 (70.6%) at enrollment (p = 0.92). Target doses of CDMT were achieved for 27% of males on ACEi/ARB/ARNI, 28% on BB, and 23% on MRA. Initial analysis of a prospective registry of males with DMD showed that ~ 30% of patients with moderate or severe LV dysfunction were not on CDMT at most recent follow-up and the majority did not reach target dosing. Further understanding regarding the optimal combination of cardiac medications for DMD cardiomyopathy is needed, as is a better understanding of the barriers to CDMT optimization given increasing cardiac causes of death for DMD patients.
期刊介绍:
The editor of Pediatric Cardiology welcomes original manuscripts concerning all aspects of heart disease in infants, children, and adolescents, including embryology and anatomy, physiology and pharmacology, biochemistry, pathology, genetics, radiology, clinical aspects, investigative cardiology, electrophysiology and echocardiography, and cardiac surgery. Articles which may include original articles, review articles, letters to the editor etc., must be written in English and must be submitted solely to Pediatric Cardiology.