Hostname: page-component-848d4c4894-ndmmz Total loading time: 0 Render date: 2024-06-12T22:58:06.306Z Has data issue: false hasContentIssue false
  • English
  • Français

Intravenous immunoglobulins in children with new onset dilated cardiomyopathy

Published online by Cambridge University Press:  11 August 2017

Josephine F. Heidendael ,
Suzanne L. Den Boer ,
Joanne G. Wildenbeest ,
Michiel Dalinghaus ,
Bart Straver  and
Dasja Pajkrt
Josephine F. Heidendael*
Affiliation:
Department of Cardiology, VU University Medical Center, Amsterdam, The Netherlands
Suzanne L. Den Boer
Affiliation:
Department of Pediatric Cardiology, Erasmus Medical Center, Sophia Children’s Hospital, Rotterdam, The Netherlands
Joanne G. Wildenbeest
Affiliation:
Department of Pediatric Infectious Diseases, Wilhelmina Children’s Hospital, University Medical Center, Utrecht, The Netherlands
Michiel Dalinghaus
Affiliation:
Department of Pediatric Cardiology, Erasmus Medical Center, Sophia Children’s Hospital, Rotterdam, The Netherlands
Bart Straver
Affiliation:
Department of Pediatric Cardiology, Academic Medical Center, Emma Children’s Hospital, Amsterdam, The Netherlands
Dasja Pajkrt
Affiliation:
Department of Pediatric Infectious Diseases, Academic Medical Center, Emma Children’s Hospital, Amsterdam, The Netherlands
*
Correspondence to: J.F. Heidendael, MD, Department of Cardiology, VU University Medical Center, De Boelelaan 1105, 1081 HV Amsterdam, The Netherlands. Tel: +31 20 444 0123; Fax: +31 20 444 2446; E-mail: j.heidendael@vumc.nl.
Get access Rights & Permissions [Opens in a new window]

Abstract

Background

Dilated cardiomyopathy is a rare but serious disorder in children. No effective diagnostic or treatment tools are readily available. This study aimed to evaluate the efficacy of intravenous immunoglobulins in children with new onset dilated cardiomyopathy.

Methods and results

In this retrospective cohort study, 94 children with new onset dilated cardiomyopathy were followed during a median period of 33 months. All patients with secondary dilated cardiomyopathy – for example, genetic, auto-immune or structural defects – had been excluded. Viral tests were performed in all patients and 18 (19%) children met the criteria for the diagnosis “probable or definite viral myocarditis”. Intravenous immunoglobulins were administered to 21 (22%) patients. Overall transplant-free survival was 75% in 5 years and did not differ between treatment groups. The treatment was associated with a higher recovery rate within 5 years, compared with non-treated children (70 versus 43%, log rank=0.045). After correction for possible confounders the hazard ratio for recovery with intravenous immunoglobulins was not significant (hazard ratio: 2.1; 95% CI: 1.0–4.6; p=0.056). Administration of intravenous immunoglobulins resulted in a greater improvement in the shortening fraction of the left ventricle.

Conclusion

In our population of children with new onset dilated cardiomyopathy, of either viral or idiopathic origin, intravenous immunoglobulins were administered to a minority of the patients and did not influence transplant-free survival, but were associated with better improvement of systolic left ventricular function and with better recovery. Our results support the concept that children with new onset dilated cardiomyopathy might benefit from intravenous immunoglobulins.

Keywords

Dilated cardiomyopathymyocarditisintravenous immunoglobulinspaediatrics
Type
Original Articles
Information
Cardiology in the Young , Volume 28 , Issue 1 , January 2018 , pp. 46 - 54
Check for updates
Copyright
© Cambridge University Press 2017 

Access options

Get access to the full version of this content by using one of the access options below. (Log in options will check for institutional or personal access. Content may require purchase if you do not have access.)

References

1. Wilkinson, JD, Landy, DC, Colan, SD, et al. The pediatric cardiomyopathy registry and heart failure: key results from the first 15 years. Heart Fail. Clin 2010; 6: 401–413.CrossRefGoogle ScholarPubMed
2. Lipshultz, SE, Cochran, TR, Briston, DA, et al. Pediatric cardiomyopathies: causes, epidemiology, clinical course, preventive strategies and therapies. Future Cardiol 2013; 9: 817–848.CrossRefGoogle ScholarPubMed
3. Felker, GM, Thompson, RE, Hare, JM, et al. Underlying causes and long-term survival in patients with initially unexplained cardiomyopathy. N Engl J Med 2000; 342: 1077–1084.CrossRefGoogle ScholarPubMed
4. Foerster, SR, Canter, CE, Cinar, A, et al. Ventricular remodeling and survival are more favorable for myocarditis than for idiopathic dilated cardiomyopathy in childhood: an outcomes study from the pediatric cardiomyopathy registry. Circ Hear Fail 2010; 3: 689–697.CrossRefGoogle ScholarPubMed
5. Klugman, D, Berger, JT, Sable, CA, He, J, Khandelwal, SG, Slonim, AD. Pediatric patients hospitalized with myocarditis: a multi-institutional analysis. Pediatr Cardiol 2010; 31: 222–228.CrossRefGoogle ScholarPubMed
6. Freund, MW, Kleinveld, G, Krediet, TG, van Loon, AM, Verboon-Maciolek, MA. Prognosis for neonates with enterovirus myocarditis. Arch Dis Child Fetal Neonatal Ed 2010; 95: F206–F212.CrossRefGoogle ScholarPubMed
7. English, RF, Janosky, JE, Ettedgui, J, Webber, S. Outcomes for children with acute myocarditis. Cardiol Young 2004; 14: 488–493.CrossRefGoogle ScholarPubMed
8. Lee, KJ, McCrindle, BW, Bohn, DJ, et al. Clinical outcomes of acute myocarditis in childhood. Heart 1999; 82: 226–233.CrossRefGoogle ScholarPubMed
9. Kaski, JP, Burch, M. Viral myocarditis in childhood. Paediatr Child Health (Oxford) 2007; 17: 11–18.CrossRefGoogle Scholar
10. Martin, B, Webber, S, Fricker, FJ, et al. Acute myocarditis. Rapid diagnosis by PCR in children. Circulation 1994; 90: 330–339.CrossRefGoogle ScholarPubMed
11. Sagar, S, Liu, PP, Cooper, LT. Myocarditis. Lancet 2012; 379: 738–747.CrossRefGoogle ScholarPubMed
12. Pophal, SG, Sigfusson, G, Booth, KL, et al. Complications of endomyocardial biopsy in children. J Am Coll Cardiol 1999; 34: 2105–2110.CrossRefGoogle ScholarPubMed
13. Friedrich, MG, Sechtem, U, Schulz-Menger, J, et al. Cardiovascular magnetic resonance in myocarditis: a JACC white paper. J Am Coll Cardiol 2009; 53: 1475–1487.CrossRefGoogle ScholarPubMed
14. Brunetti, L, DeSantis, ERH. Treatment of viral myocarditis caused by coxsackievirus B. Am J Health Syst Pharm 2008; 65: 132–137.CrossRefGoogle ScholarPubMed
15. Mason, JW, O’Connell, JB, Herskowitz, A, et al. A clinical trial of immunosuppressive therapy for myocarditis. The myocarditis treatment trial investigators. N Engl J Med 1995; 333: 269–275.CrossRefGoogle ScholarPubMed
16. Ikeda, U, Kasai, H, Izawa, A, et al. Immunoadsorption therapy for patients with dilated cardiomyopathy and heart failure. Curr Cardiol Rev 2008; 4: 219–222.CrossRefGoogle ScholarPubMed
17. Maisch, B, Hufnagel, G, Kölsch, S, et al. Treatment of inflammatory dilated cardiomyopathy and (peri)myocarditis with immunosuppression and i.v. immunoglobulins. Herz 2004; 29: 624–636.CrossRefGoogle ScholarPubMed
18. McNamara, DM, Holubkov, R, Starling, RC, et al. Controlled trial of intravenous immune globulin in recent-onset dilated cardiomyopathy. Circulation 2001; 103: 2254–2259.CrossRefGoogle ScholarPubMed
19. Bhatt, GC, Sankar, J, Kushwaha, KP. Use of intravenous immunoglobulin compared with standard therapy is associated with improved clinical outcomes in children with acute encephalitis syndrome complicated by myocarditis. Pediatr Cardiol 2012; 33: 1370–1376.CrossRefGoogle ScholarPubMed
20. Haque, A, Bhatti, S, Siddiqui, FJ. Intravenous immune globulin for severe acute myocarditis in children. Indian Pediatr 2009; 46: 810–811.Google ScholarPubMed
21. Drucker, N, Colan, SD, Lewis, B, et al. Gamma-globulin treatment of acute myocarditis in the pediatric population. Circulation 1994; 89: 252–257.CrossRefGoogle ScholarPubMed
22. Prasad, AN, Chaudhary, S. Intravenous immunoglobulin in children with acute myocarditis and/or early dilated cardiomyopathy. Indian Pediatr 2014; 51: 583–584.CrossRefGoogle ScholarPubMed
23. Den Boer, SL, Meijer, RPJ, van Iperen, GG, et al. Evaluation of the diagnostic work-up in children with myocarditis and idiopathic dilated cardiomyopathy. Pediatr Cardiol 2015; 32: 409–416.CrossRefGoogle Scholar
24. Sluysmans, T, Colan, SD. Theoretical and empirical derivation of cardiovascular allometric relationships in children. J. Appl. Physiol 2005; 99: 445–457.CrossRefGoogle ScholarPubMed
25. Dennert, R, Velthuis, S, Schalla, S, et al. Intravenous immunoglobulin therapy for patients with idiopathic cardiomyopathy and endomyocardial biopsy-proven high PVB19 viral load. Antivir Ther 2010; 15: 193–201.CrossRefGoogle Scholar
26. Kishimoto, C. Treatment of acute inflammatory cardiomyopathy with intravenous immunoglobulin ameliorates left ventricular function associated with suppression of inflammatory cytokines and decreased oxidative stress. Int J Cardiol 2003; 91: 173–178.CrossRefGoogle ScholarPubMed
27. McNamara, DM, Rosenblum, WD, Janosko, KM, et al. Intravenous immune globulin in the therapy of myocarditis and acute cardiomyopathy. Circulation 1997; 95: 2476–2478.CrossRefGoogle ScholarPubMed
28. Goland, S, Czer, LSC, Siegel, RJ, et al. Intravenous immunoglobulin treatment for acute fulminant inflammatory cardiomyopathy: series of six patients and review of literature. Can J Cardiol 2008; 24: 571–574.CrossRefGoogle ScholarPubMed
29. Chen, SC. Echocardiography of dilated cardiomyopathy in children. Echocardiography 1991; 8: 207–217.CrossRefGoogle ScholarPubMed
30. Slama, M, Maizel, J. Echocardiographic measurement of ventricular function. Curr Opin Crit Care 2006; 12: 241–248.CrossRefGoogle ScholarPubMed
31. Dragulescu, A, Mertens, LL. Developments in echocardiographic techniques for the evaluation of ventricular function in children. Arch Cardiovasc Dis 2010; 103: 603–614.CrossRefGoogle ScholarPubMed
32. Caforio, ALP, Pankuweit, S, Arbustini, E, et al. Current state of knowledge on aetiology, diagnosis, management, and therapy of myocarditis: a position statement of the European Society of Cardiology Working Group on myocardial and pericardial diseases. Eur Heart J 2013; 34: 2636–2648.CrossRefGoogle Scholar
Supplementary material: File

Heidendael supplementary material

Table S1

Download Heidendael supplementary material(File)
File 17.5 KB