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• W2120718081 abstract "HomeCirculation: Arrhythmia and ElectrophysiologyVol. 4, No. 3When Should High-Grade Heart Block Trigger a Search for a Treatable Cardiomyopathy? Free AccessEditorialPDF/EPUBAboutView PDFView EPUBSections ToolsAdd to favoritesDownload citationsTrack citationsPermissions ShareShare onFacebookTwitterLinked InMendeleyReddit Jump toFree AccessEditorialPDF/EPUBWhen Should High-Grade Heart Block Trigger a Search for a Treatable Cardiomyopathy? Leslie T. CooperJr and Lori A. Blauwet Leslie T. CooperJrLeslie T. CooperJr From the Division of Cardiovascular Diseases, Mayo Clinic, Rochester, MN. Search for more papers by this author and Lori A. BlauwetLori A. Blauwet From the Division of Cardiovascular Diseases, Mayo Clinic, Rochester, MN. Search for more papers by this author Originally published1 Jun 2011https://doi.org/10.1161/CIRCEP.111.963249Circulation: Arrhythmia and Electrophysiology. 2011;4:260–261High-degree heart block is an uncommon manifestation of acute myocarditis in adults. The rate of heart block requiring a pacemaker in biopsy-proven acute lymphocytic myocarditis generally is low but has been reported in up to 8.3% of cases.1 Important exceptions include cardiac sarcoidosis and giant cell myocarditis (GCM), 2 uncommon and idiopathic disorders for which early immunosuppression may improve clinical outcomes. High-degree heart block occurs in the course of biopsy-proven cardiac sarcoidosis in 23% to 30% of cases (Table).2 In GCM, second- or third-degree heart block requiring a pacemaker occurs in 25% of cases.3Table. Prevalence of Various Cardiac Findings in Cardiac Sarcoidosis During the Course of DiseasePrevalence in Study Series, %Atrioventricular block26–62Bundle-branch block12–61Supraventricular tachycardia0–15Ventricular tachycardia2–42Congestive heart failure10–30Sudden death12–65Adapted with permission from Kim et al.2Article see p 303This relatively high rate of heart block reflects the severity of the clinical course of cardiac sarcoidosis and GCM, which have a higher rate of death and heart transplantation than lymphocytic myocarditis.1,4 Unlike acute lymphocytic myocarditis, for which immunosuppression usually is not beneficial,5 GCM or cardiac sarcoidosis may improve with certain combinations of immunosuppressive drugs. In the case of acute GCM, cyclosporine combined with corticosteroids and usually a T-cell lytic agent like muromonab-CD3 improves transplant-free survival.6 Uncontrolled case series also suggested that left ventricular (LV) dysfunction due to cardiac sarcoidosis may improve with corticosteroid therapy.7Because GCM and cardiac sarcoidosis are rare disorders, endomyocardial biopsy is not routinely recommended to detect them in the setting of complete heart block unless cardiomyopathy is present.8 Although a recommendation class was not assigned to this clinical scenario by the American Heart Association/American College of Cardiology Foundation/European Society of Cardiology (AHA/ACCF/ESC) Writing Group, the role of endomyocardial biopsy in idiopathic complete heart block was discussed in the text of the scientific statement. On the basis of expert opinion, the diagnostic yield of right ventricular septal endomyocardial biopsy in this clinical scenario would be quite low relative to the potential benefits. However, the frequency of specific and treatable disorders among young patients who present with heart block requiring a pacemaker only has been reported in a single Japanese case series9 until now.In this issue of Circulation: Arrhythmia & Electrophysiology, Kandolin et al10 report a case series of 72 young adult patients aged 18 to 55 years with initially unexplained atrioventricular block in whom cardiac sarcoidosis or GCM was found in 14 (19%) and 4 (6%) patients, respectively. Consistent with prior reports, none of the patients had lymphocytic myocarditis. Over an average follow-up of 48 months, 7 (39%) of the 18 patients with cardiac sarcoidosis or GCM experienced cardiac death, cardiac transplantation, or ventricular fibrillation or were treated for sustained ventricular tachycardia. This rate compares to only 1 (2%) of the 54 patients in whom atrioventricular block remained idiopathic (P<0.001). This report is the first case series in which the rate of GCM and cardiac sarcoidosis is reported in a consecutive series of patients requiring pacemaker implantation.Several aspects of the Kandolin et al study10 deserve comment. To what degree are their results generalizable to other adult populations with atrioventricular block? The authors' study population of young adults without clinically apparent causes for heart block represents a small fraction of the 6420 persons who received pacemakers during the 10-year study period. Only 212 (3.3%) were aged <55 years, whereas just slightly >1% (72/6420) had high-grade idiopathic heart block. The rate of cardiac sarcoidosis and GCM in an older cohort with established reasons for heart block certainly would be much lower.The study cohort was from Finland, a country with a relatively high background rate of systemic sarcoidosis.11 It is not known whether populations with different ethnic backgrounds and lower rates of systemic sarcoidosis would have similar findings. Most cases of GCM are described in whites, but this observation may reflect biases in diagnosis rate or variable presentations rather than a true difference in GCM incidence.4 In populations of African or Asian ancestry who have unexplained cardiomyopathy or heart block, the rates of cardiac biopsy-proven GCM or cardiac sarcoidosis are not known.To identify the 18 cases of histologically proven GCM and cardiac sarcoidosis, the authors searched systematically for a specific cause of cardiomyopathy.10 Cardiac MRI, PET, and endomyocardial biopsy commonly were used. The 36 endomyocardial biopsy specimens (5 to 12 samples per session) of the LV, right ventricle, or both in 25 of 72 patients yielded 9 positive cases. Six cases of cardiac sarcoidosis and 1 case of GCM were diagnosed on initial biopsy, and 1 cardiac sarcoidosis case and 1 GCM case were diagnosed on second biopsy. This notably high rate of specific cardiomyopathy detection may reflect more diffuse myocardial disease at the time of heart block diagnosis. Indeed, the mean LV ejection fraction in the final cohort with unexplained heart block was mildly reduced at 50% (range, 25% to 70%), suggesting that about half of the cases had clear evidence of LV systolic dysfunction at the time of diagnosis. Interestingly, the 25% (9 of 36) rate of positive biopsy specimens is similar to the 28% (8 of 28) positive rate in a prospective GCM registry in which the criteria for biopsy were an acute cardiomyopathy complicated by heart block, ventricular tachycardia, or failure to respond to usual care.6The optimal initial and follow-up imaging strategy for patients with suspected GCM or cardiac sarcoidosis is not known. Several reports suggest that cardiac MRI may have specific features for cardiac sarcoidosis.12,13 The diagnosis of sarcoidosis by 18-fluorodeoxyglucose PET is less well established but, not infrequently, can identify a site of extracardiac sarcoidosis for biopsy.14 Endomyocardial voltage mapping has been used to guide biopsy to regions of low voltage in suspected cardiac sarcoidosis. Greater use of this and similar technologies may increase the sensitivity of biopsy, particularly in a disease with focal myocardial involvement like cardiac sarcoidosis. In those patients with normal cardiac function at the time of presentation with heart block, it would be reasonable to repeat an echocardiogram 6 to 12 months later to monitor for development of cardiomyopathy.Should these findings affect the current AHA/ACCF/ESC scientific statement for use of endomyocardial biopsy? The 25% positive biopsy rate is high enough that endomyocardial biopsy should probably be recommended in populations at similar risk (based on ethnicity, age, and exclusion of more common causes). In select cases, the recommendation for endomyocardial biopsy probably should be class 2A (a reasonable procedure). However, to minimize risk and maximize the likelihood of a diagnostic sample, the medical center and operator should have experience in the procedure, as manifested by a low procedural complication rate, and timely and expert cardiac pathology consultation should be available.From this and other studies, we know that a histological diagnosis of acute GCM will affect management and outcome. In the setting of cardiomyopathy, a biopsy-based diagnosis of sarcoidosis carries a poor prognosis.15 However, a remaining gap in our knowledge of cardiac sarcoidosis is whether treatment changes based on a histological diagnosis, or a probable diagnosis based on imaging abnormalities, will affect clinically meaningful outcomes in the clinical scenario of isolated heart block. Is the likelihood of recovery of atrioventricular conduction greater or risk of dilated cardiomyopathy lower with immunosuppressive therapy? In the Kandolin et al10 study, there was a trend toward less pacing on follow-up in the early immunosuppression group (P=0.082), but larger, multicenter prospective registries and trials are needed to adequately address the effect of a specific diagnosis-guided treatment on outcomes.DisclosuresNone.FootnotesThe opinions expressed in this article are not necessarily those of the editors or of the American Heart Association.Correspondence to Leslie T. Cooper, Jr, MD, Division of Cardiovascular Diseases, Mayo Clinic, 200 First St SW, Rochester, MN 55905. E-mail cooper.[email protected]eduReferences1. Davidoff R, Palacios I, Southern J, Fallon JT, Newell J, Dec GW. Giant cell versus lymphocytic myocarditis: a comparison of their clinical features and long-term outcomes. Circulation. 1991; 83:953–961.LinkGoogle Scholar2. Kim JS, Judson MA, Donnino R, Gold M, Cooper LT, Prystowsky EN, Prystowsky S. Cardiac sarcoidosis. Am Heart J. 2009; 157:9–21.CrossrefMedlineGoogle Scholar3. Okura Y, Dec GW, Hare JM, Kodama M, Berry GJ, Tazelaar HD, Bailey KR, Cooper LT. A clinical and histopathologic comparison of cardiac sarcoidosis and idiopathic giant cell myocarditis. J Am Coll Cardiol. 2003; 41:322–329.CrossrefMedlineGoogle Scholar4. Cooper LT, Berry GJ, Shabetai R; Multicenter Giant Cell Myocarditis Study Group Investigators. Idiopathic giant-cell myocarditis—natural history and treatment. N Engl J Med. 1997; 336:1860–1866.CrossrefMedlineGoogle Scholar5. Mason JW, O'Connell JB, Herskowitz A, Rose NR, McManus BM, Billingham ME, Moon TE; Myocarditis Treatment Trial Investigators. A clinical trial of immunosuppressive therapy for myocarditis. N Engl J Med. 1995; 333:269–275.CrossrefMedlineGoogle Scholar6. Cooper LT, Hare JM, Tazelaar HD, Edwards WD, Starling RC, Deng MC, Menon S, Mullen GM, Jaski B, Bailey KR, Cunningham MW, Dec GW; Giant Cell Myocarditis Treatment Trial Investigators. Usefulness of immunosuppression for giant cell myocarditis. Am J Cardiol. 2008; 102:1535–1539.CrossrefMedlineGoogle Scholar7. Yazaki Y, Isobe M, Hiroe M, Morimoto S, Hiramitsu S, Nakano T, Izumi T, Sekiguchi M. Central Japan Heart Study Group. Prognostic determinants of long-term survival in Japanese patients with cardiac sarcoidosis treated with prednisone. Am J Cardiol. 2001; 88:1006–1010.CrossrefMedlineGoogle Scholar8. Cooper LT, Baughman KL, Feldman AM, Frustaci A, Jessup M, Kuhl U, Levine GN, Narula J, Starling RC, Towbin J, Virmani R; American Heart Association; American College of Cardiology; European Society of Cardiology; Heart Failure Society of America; Heart Failure Association of the European Society of Cardiology. The role of endomyocardial biopsy in the management of cardiovascular disease: a scientific statement from the American Heart Association, the American College of Cardiology, and the European Society of Cardiology. Circulation. 2007; 116:2216–2233.LinkGoogle Scholar9. Uemura A, Morimoto S, Hiramitsu S, Hishida H. Endomyocardial biopsy findings in 50 patients with idiopathic atrioventricular block: presence of myocarditis. Jpn Heart J. 2001; 42:691–700.CrossrefMedlineGoogle Scholar10. Kandolin R, Lehtonen J, Kupari M. Cardiac sarcoidosis and giant cell myocarditis as causes of atrioventricular block in young and middle-aged adults. Circ Arrhythm Electrophysiol. 2011; 4:303–309.LinkGoogle Scholar11. Bresnitz EA, Strom BL. Epidemiology of sarcoidosis. Epidemiol Rev. 1983; 5:124–156.CrossrefMedlineGoogle Scholar12. Schulz-Menger J, Wassmuth R, Abdel-Aty H, Siegel I, Franke A, Dietz R, Friedrich MG. Patterns of myocardial inflammation and scarring in sarcoidosis as assessed by cardiovascular magnetic resonance. Heart. 2006; 92:399–400.CrossrefMedlineGoogle Scholar13. Vignaux O. Cardiac sarcoidosis: spectrum of MRI features. AJR Am J Roentgenol. 2005; 184:249–254.CrossrefMedlineGoogle Scholar14. Okumura W, Iwasaki T, Toyama T, Iso T, Arai M, Oriuchi N, Endo K, Yokoyama T, Suzuki T, Kurabayashi M. Usefulness of fasting 18F-FDG PET in identification of cardiac sarcoidosis. J Nucl Med. 2004; 45:989–998.Google Scholar15. Ardehali H, Howard DL, Hariri A, Qasim A, Hare JM, Baughman KL, Kasper EK. A positive endomyocardial biopsy result for sarcoid is associated with poor prognosis in patients with initially unexplained cardiomyopathy. Am Heart J. 2005; 150:459–463.CrossrefMedlineGoogle Scholar Previous Back to top Next FiguresReferencesRelatedDetailsCited By Alblaihed L, Brady W, Al-Salamah T and Mattu A (2023) Dysrhythmias associated with COVID-19: Review and management considerations, The American Journal of Emergency Medicine, 10.1016/j.ajem.2022.12.004, 64, (161-168), Online publication date: 1-Feb-2023. Desai A, Boursiquot B, Melki L and Wan E (2020) Management of Arrhythmias Associated with COVID-19, Current Cardiology Reports, 10.1007/s11886-020-01434-7, 23:1, Online publication date: 1-Jan-2021. Kir D, Mohan C and Sancassani R (2020) Heart Brake, JACC: Case Reports, 10.1016/j.jaccas.2020.04.026, 2:9, (1252-1255), Online publication date: 1-Jul-2020. Hozman M and Schee A (2018) Intermittent high-grade atrioventricular block as a rare extra-articular manifestation of rheumatoid arthritis, Intervenční a akutní kardiologie, 10.36290/kar.2018.059, 17:4, (225-227), Online publication date: 1-Dec-2018. Patel B, Shah M, Gelaye A and Dusaj R (2016) A complete heart block in a young male: a case report and review of literature of cardiac sarcoidosis, Heart Failure Reviews, 10.1007/s10741-016-9585-0, 22:1, (55-64), Online publication date: 1-Jan-2017. Rosenthal D, Bravo P, Patton K and Goldberger Z (2015) Management of Arrhythmias in Cardiac Sarcoidosis, Clinical Cardiology, 10.1002/clc.22430, 38:10, (635-640), Online publication date: 1-Oct-2015. Liang J, Hebl V, DeSimone C, Madhavan M, Nanda S, Kapa S, Maleszewski J, Edwards W, Reeder G, Cooper L and Asirvatham S (2014) Electrogram Guidance, JACC: Heart Failure, 10.1016/j.jchf.2014.03.015, 2:5, (466-473), Online publication date: 1-Oct-2014. NERY P, BEANLANDS R, NAIR G, GREEN M, YANG J, MCARDLE B, DAVIS D, OHIRA H, GOLLOB M, LEUNG E, HEALEY J and BIRNIE D (2014) Atrioventricular Block as the Initial Manifestation of Cardiac Sarcoidosis in Middle-Aged Adults, Journal of Cardiovascular Electrophysiology, 10.1111/jce.12401, 25:8, (875-881), Online publication date: 1-Aug-2014. NERY P, Mc ARDLE B, REDPATH C, LEUNG E, LEMERY R, DEKEMP R, YANG J, KEREN A, BEANLANDS R and BIRNIE D (2013) Prevalence of Cardiac Sarcoidosis in Patients Presenting with Monomorphic Ventricular Tachycardia, Pacing and Clinical Electrophysiology, 10.1111/pace.12277, 37:3, (364-374), Online publication date: 1-Mar-2014. Veia A, Cavallino C, Bacchini S, Pastore F, Lupi A, Rognoni A, Rametta F and Bongo A (2014) Idiopathic Giant Cell Myocarditis: State of the Art, World Journal of Cardiovascular Diseases, 10.4236/wjcd.2014.46041, 04:06, (316-324), . Blauwet L and Cooper L (2012) Idiopathic giant cell myocarditis and cardiac sarcoidosis, Heart Failure Reviews, 10.1007/s10741-012-9358-3, 18:6, (733-746), Online publication date: 1-Nov-2013. June 2011Vol 4, Issue 3 Advertisement Article InformationMetrics © 2011 American Heart Association, Inc.https://doi.org/10.1161/CIRCEP.111.963249PMID: 21673024 Originally publishedJune 1, 2011 Keywordsmyocarditisgiant cellscardiomyopathyEditorialssarcoidosisheart blockPDF download Advertisement SubjectsArrhythmiasCardiomyopathyHeart Failure" @default.
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