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European Heart Journal Advance Access published online on July 3, 2006
European Heart Journal, doi:10.1093/eurheartj/ehl112
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European Heart Journal © The European Society of Cardiology 2006; all rights reserved
Received September 28, 2004
Revised April 5, 2006
Accepted June 1, 2006

Clinical research

Complications of non-compaction of the left ventricular myocardium in a paediatric population: a prospective study

Christian Lilje 1 *, Vit Rázek 2, James J. Joyce 3, Thomas Rau 4, Barbara F. Finckh 5, Florian Weiss 6, Christian R. Habermann 6, Janet C. Rice 7, and Jochen Weil 2

1 Departments of Kinderkardiologie, Universitäts-Klinikum Hamburg-Eppendorf, Martinistr. 52, D-20246 Hamburg, Germany; Section of Pediatric Cardiology, Tulane University Health Sciences Center, New Orleans, LA, USA
2 Departments of Kinderkardiologie, Universitäts-Klinikum Hamburg-Eppendorf, Martinistr. 52, D-20246 Hamburg, Germany
3 Section of Pediatric Cardiology, Tulane University Health Sciences Center, New Orleans, LA, USA
4 Experimentelle & Klinische Pharmakologie & Toxikologie, Universitäts-Klinikum Hamburg-Eppendorf, Hamburg, Germany
5 Clinical Pathology/Pediatrics, Universitäts-Klinikum Hamburg-Eppendorf, Hamburg, Germany
6 Diagnostische Radiologie, Universitäts-Klinikum Hamburg-Eppendorf, Hamburg, Germany
7 Department of Biostatistics, Tulane University School of Public Health & Tropical Medicine, New Orleans, LA, USA

* To whom correspondence should be addressed.
Christian Lilje, E-mail: lilje{at}europe.com


  Abstract

Aims Non-compaction of the left ventricular myocardium (NCVM) is reportedly exceedingly rare and associated with a high morbidity and mortality. A different genetic background has been suggested for NCVM with [non-isolated NCVM (ni-NCVM)] and without [isolated NCVM (i-NCVM)] other congenital heart defects. We prospectively evaluated both the NCVM subgroups regarding frequency of occurrence and cardiovascular complications in a paediatric population.

Results In a prospective, single-centre study, 66/5220 consecutive patients (1.26%) were diagnosed (25 i-NCVM, 41 ni-NCVM). The median age was 4 years (range 0-21), the median follow-up 12 months (range 0-51). The occurrence of congestive heart failure (CHF) at follow-up was 68.0%. CHF was as frequently seen in i-NCVM and ni-NCVM patients (77.5 vs. 62.1%, P = 0.322). The occurrence of arrhythmias (20.0%) and thrombo-embolic events (13.9%) was not different between subgroups. The cardiomyopathy related mortality was 7.1%, with three and one deaths in the i-NCVM and ni-NCVM groups, respectively (P = 0.126).

Conclusion When prospectively evaluated, NCVM appears to have been previously under-diagnosed. Whereas arrhythmias and thrombo-embolic events were rare, CHF was frequently found. An equally aggressive anticongestive treatment regimen would seem indicated for both the NCVM subgroups.

Keywords: Heart defects, congenital; Cardiomyopathy; Heart failure, congestive; Echocardiography; Non-compaction.
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