{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,10,8]],"date-time":"2026-10-08T12:28:26Z","timestamp":1791462506528,"version":"4.3.4"},"reference-count":28,"publisher":"Ovid Technologies (Wolters Kluwer Health)","issue":"14","content-domain":{"domain":["www.ahajournals.org"],"crossmark-restriction":true},"short-container-title":["Circulation"],"published-print":{"date-parts":[[2025,10,7]]},"abstract":"<jats:sec>\n                    <jats:title>BACKGROUND:<\/jats:title>\n                    <jats:p>Inflammatory, myocarditis-like episodes precede and are associated with higher risk of sustained ventricular arrhythmias and heart failure in patients with pathogenic or likely pathogenic desmoplakin (DSP) variants. Whether the recurrence and treatment of myocarditis-like episodes influence the outcomes in this population is unknown. This study aimed to assess the prognostic impact of the recurrence and treatment of myocarditis-like episodes in patients with pathogenic or likely pathogenic DSP variants.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>METHODS:<\/jats:title>\n                    <jats:p>\n                      The study was designed as an observational cohort study using all patients with pathogenic or likely pathogenic DSP variants in the\n                      <jats:italic toggle=\"yes\">DSP<\/jats:italic>\n                      -ERADOS Network (\n                      <jats:italic toggle=\"yes\">Desmopakin<\/jats:italic>\n                      Specific Effort for a Rare Disease Outcome Study) enrolled at 31 institutions up until April 30, 2024. Survival from ventricular arrhythmia and heart failure end points and recurrent myocarditis-like episodes was evaluated for both first and recurrent documented myocarditis-like episodes and the impact of immunosuppressive treatment on outcomes assessed.\n                    <\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>RESULTS:<\/jats:title>\n                    <jats:p>\n                      A total of 1014 patients with pathogenic or likely pathogenic DSP variants (62.4% female; mean age, 40.4\u00b117.4 years; 47.1% probands) were included. Among them, 177 individuals (17.5%) experienced \u22651 documented myocarditis-like episode, with 63 of them (35.6%) receiving immunosuppressive treatment. Immunosuppressive treatment of the first documented myocarditis-like episode was associated with reduced risk of ventricular arrhythmia and heart failure outcomes (hazard ratio, 0.18 [95% CI, 0.07\u20130.45];\n                      <jats:italic toggle=\"yes\">P<\/jats:italic>\n                      &lt;0.001; and hazard ratio, 0.09 [95% CI, 0.02\u20130.39;\n                      <jats:italic toggle=\"yes\">P<\/jats:italic>\n                      =0.001, respectively) over a median follow-up of 6.4 years (95% CI, 4.0\u201310.5). This risk was comparable to that of patients with no history of myocarditis-like episode but did not correlate with a reduction in recurrent episodes after completion of initial immunosuppression (hazard ratio, 0.85 [95% CI, 0.47\u20131.54];\n                      <jats:italic toggle=\"yes\">P<\/jats:italic>\n                      =0.599). In contrast, nonsteroidal anti-inflammatory drugs or colchicine treatment did not influence outcomes.\n                    <\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>CONCLUSIONS:<\/jats:title>\n                    <jats:p>\n                      Immunosuppressive treatment of myocarditis-like episodes in\n                      <jats:italic toggle=\"yes\">DSP<\/jats:italic>\n                      patients was associated with improved combined ventricular arrhythmia or heart failure outcomes, supporting future prospective evaluation in DSP cohorts.\n                    <\/jats:p>\n                  <\/jats:sec>","DOI":"10.1161\/circulationaha.125.073919","type":"journal-article","created":{"date-parts":[[2025,8,20]],"date-time":"2025-08-20T09:00:21Z","timestamp":1755680421000},"page":"978-989","update-policy":"https:\/\/doi.org\/10.1161\/crossmarkpolicy","source":"Crossref","is-referenced-by-count":39,"title":["Prognostic Role of Myocarditis-Like Episodes and Their Treatment in Patients With Pathogenic Desmoplakin Variants"],"prefix":"10.1161","volume":"152","author":[{"ORCID":"https:\/\/orcid.org\/0000-0003-3432-070X","authenticated-orcid":true,"given":"Alessio","family":"Gasperetti","sequence":"first","affiliation":[{"name":"Department of Medicine, Division of Cardiology, Johns Hopkins University School of Medicine, Baltimore, MD (A.G., S.A.M., B.A., R.T.C., B.M., C. Tichnell, P.S., L.A., H.C., C.A.J., N.A.G.)."}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-2674-8709","authenticated-orcid":true,"given":"Steven A.","family":"Muller","sequence":"additional","affiliation":[{"name":"Department of Medicine, Division of Cardiology, Johns Hopkins University School of Medicine, Baltimore, MD (A.G., S.A.M., B.A., R.T.C., B.M., C. Tichnell, P.S., L.A., H.C., C.A.J., N.A.G.)."},{"name":"Division of Cardiology, University Medical Center Utrecht, the Netherlands (S.A.M., J.P.v.T., A.S.J.M.t.R.)."}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-1815-4000","authenticated-orcid":true,"given":"Giovanni","family":"Peretto","sequence":"additional","affiliation":[{"name":"Multidisciplinary Disease Unit for Myocarditis and Arrhythmogenic Cardiomyopathies, IRCCS San Raffaele Scientific Institute, Milan, Italy (G.P.)."}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-0050-5717","authenticated-orcid":true,"given":"Babken","family":"Asatryan","sequence":"additional","affiliation":[{"name":"Department of Medicine, Division of Cardiology, Johns Hopkins University School of Medicine, Baltimore, MD (A.G., S.A.M., B.A., R.T.C., B.M., C. Tichnell, P.S., L.A., H.C., C.A.J., N.A.G.)."}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Alexandros","family":"Protonotarios","sequence":"additional","affiliation":[{"name":"Inherited Cardiovascular Diseases Unit, St Bartholomew\u2019s Hospital, UCL Institute of Cardiovascular Science, London, UK (A.P., P.S., P.M.E.)."}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-7326-2656","authenticated-orcid":true,"given":"Mikael","family":"Laredo","sequence":"additional","affiliation":[{"name":"Sorbonne Universit\u00e9, AP-HP, IHU-ICAN, Groupe Hospitalier Piti\u00e9-Salp\u00eatri\u00e8re, Institut de Cardiologie, Paris, France (M.L., P.-Y.T., P.C., F.A., E.G.)."},{"name":"University of Colorado Cardiovascular Institute, University of Colorado Anschutz Medical Campus, Aurora (K.M., M.J., L.M., M.T.)."}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Pierre-Yves","family":"Tarlet","sequence":"additional","affiliation":[{"name":"Sorbonne Universit\u00e9, AP-HP, IHU-ICAN, Groupe Hospitalier Piti\u00e9-Salp\u00eatri\u00e8re, Institut de Cardiologie, Paris, France (M.L., P.-Y.T., P.C., F.A., E.G.)."}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-2363-8758","authenticated-orcid":true,"given":"Petros","family":"Syrris","sequence":"additional","affiliation":[{"name":"Inherited Cardiovascular Diseases Unit, St Bartholomew\u2019s Hospital, UCL Institute of Cardiovascular Science, London, UK (A.P., P.S., P.M.E.)."}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-5589-1216","authenticated-orcid":true,"given":"Richard T.","family":"Carrick","sequence":"additional","affiliation":[{"name":"Department of Medicine, Division of Cardiology, Johns Hopkins University School of Medicine, Baltimore, MD (A.G., S.A.M., B.A., R.T.C., B.M., C. Tichnell, P.S., L.A., H.C., C.A.J., N.A.G.)."}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Brittney","family":"Murray","sequence":"additional","affiliation":[{"name":"Department of Medicine, Division of Cardiology, Johns Hopkins University School of Medicine, Baltimore, MD (A.G., S.A.M., B.A., R.T.C., B.M., C. Tichnell, P.S., L.A., H.C., C.A.J., N.A.G.)."}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Crystal","family":"Tichnell","sequence":"additional","affiliation":[{"name":"Department of Medicine, Division of Cardiology, Johns Hopkins University School of Medicine, Baltimore, MD (A.G., S.A.M., B.A., R.T.C., B.M., C. Tichnell, P.S., L.A., H.C., C.A.J., N.A.G.)."}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Paul","family":"Scheel","sequence":"additional","affiliation":[{"name":"Department of Medicine, Division of Cardiology, Johns Hopkins University School of Medicine, Baltimore, MD (A.G., S.A.M., B.A., R.T.C., B.M., C. Tichnell, P.S., L.A., H.C., C.A.J., N.A.G.)."}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Marta","family":"Gigli","sequence":"additional","affiliation":[{"name":"Cardiothoracovascular Department, Division of Cardiology, Azienda Sanitaria Universitaria Giuliano Isontina and University of Trieste, Italy (M.G., G.S., M.M.)."}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-4520-2725","authenticated-orcid":true,"given":"Sing-Chien","family":"Yap","sequence":"additional","affiliation":[{"name":"Department of Cardiology, Thorax Center, Cardiovascular Institute, Erasmus MC, Rotterdam, the Netherlands (S.-C.Y.)."}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-3646-0669","authenticated-orcid":true,"given":"Kalliopi","family":"Pilichou","sequence":"additional","affiliation":[{"name":"Department of Cardiac, Thoracic, Vascular Sciences and Public Health, University of Padua, Italy (K.P., C.B., B.B.)."}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-0195-9753","authenticated-orcid":true,"given":"Cristina","family":"Basso","sequence":"additional","affiliation":[{"name":"Department of Cardiac, Thoracic, Vascular Sciences and Public Health, University of Padua, Italy (K.P., C.B., B.B.)."}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0009-0007-9785-4668","authenticated-orcid":true,"given":"Kristen","family":"Medo","sequence":"additional","affiliation":[{"name":"University of Colorado Cardiovascular Institute, University of Colorado Anschutz Medical Campus, Aurora (K.M., M.J., L.M., M.T.)."}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0009-0003-1492-2276","authenticated-orcid":true,"given":"Max","family":"Jason","sequence":"additional","affiliation":[{"name":"University of Colorado Cardiovascular Institute, University of Colorado Anschutz Medical Campus, Aurora (K.M., M.J., L.M., M.T.)."}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-9235-6597","authenticated-orcid":true,"given":"Valentina A.","family":"Rossi","sequence":"additional","affiliation":[{"name":"Department of Cardiology, Arrhythmia Unit, University Heart Center, University Hospital Zurich, Switzerland (V.A.R.., A.M.S., F.D.)."}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-1896-0803","authenticated-orcid":true,"given":"Ardan M.","family":"Saguner","sequence":"additional","affiliation":[{"name":"Department of Cardiology, Arrhythmia Unit, University Heart Center, University Hospital Zurich, Switzerland (V.A.R.., A.M.S., F.D.)."}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-4748-0158","authenticated-orcid":true,"given":"Firat","family":"Duru","sequence":"additional","affiliation":[{"name":"Department of Cardiology, 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of Colorado Cardiovascular Institute, University of Colorado Anschutz Medical Campus, Aurora (K.M., M.J., L.M., M.T.)."},{"name":"Department of Experimental and Clinical Medicine, University of Florence, Meyer Children Hospital and Careggi University Hospital, Italy (M.T., M.P.)."}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-6830-4292","authenticated-orcid":true,"given":"Maurizio","family":"Pieroni","sequence":"additional","affiliation":[{"name":"Department of Experimental and Clinical Medicine, University of Florence, Meyer Children Hospital and Careggi University Hospital, Italy (M.T., M.P.)."},{"name":"Department of Cardiology, Arezzo University Hospital, Italy (M.P.)."}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Maddalena","family":"Graziosi","sequence":"additional","affiliation":[{"name":"Cardiac Thoracic and Vascular Department, Cardiology Unit, IRCCS Azienda Ospedaliero-Universitaria di Bologna, Italy (M.G., 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Cox)."}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-5322-1742","authenticated-orcid":true,"given":"Michela","family":"Casella","sequence":"additional","affiliation":[{"name":"Cardiology and Arrhythmology Clinic, University Hospital \u201cOspedali Riuniti,\u201d Department of Clinical, Special and Dental Sciences, Marche Polytechnic University, Ancona, Italy (M. Casella)."}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-8500-8313","authenticated-orcid":true,"given":"Claudio","family":"Tondo","sequence":"additional","affiliation":[{"name":"Centro Cardiologico Monzino, IRCCS, Department of Clinical Electrophysiology and Cardiac Pacing, Milan, Italy (C. Tondo)."}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-6110-5880","authenticated-orcid":true,"given":"James S.","family":"Ware","sequence":"additional","affiliation":[{"name":"National Heart and Lung Institute and MRC London Institute of Medical Sciences, Imperial College London, UK (J.S.W., S.K.P.)."}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-5637-0727","authenticated-orcid":true,"given":"Sanjay K.","family":"Prasad","sequence":"additional","affiliation":[{"name":"National Heart and Lung Institute and MRC London Institute of Medical Sciences, Imperial College London, UK (J.S.W., S.K.P.)."}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-6062-5286","authenticated-orcid":true,"given":"Leonardo","family":"Cal\u00f2","sequence":"additional","affiliation":[{"name":"Department of Cardiology, Policlinico Casilino, Rome, Italy (L. 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