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Circulation, Vol 82, 507-513, Copyright © 1990 by American Heart Association
TK Chin, JK Perloff, RG Williams, K Jue and R Mohrmann
Isolated noncompaction of left ventricular myocardium is a rare disorder of
endomyocardial morphogenesis characterized by numerous, excessively
prominent ventricular trabeculations and deep intertrabecular recesses.
This study comprised eight cases, including three at necropsy. Ages ranged
from 11 months to 22.5 years, with follow-up as long as 5 years. Gross
morphological severity ranged from moderately abnormal ventricular
trabeculations to profoundly abnormal, loosely compacted trabeculations.
Echocardiographic images were diagnostic and corresponded to the
morphological appearances at necropsy. The depths of the intertrabecular
recesses were assessed by a quantitative echocardiographic X-to-Y ratio and
were significantly greater than in normal control subjects (p less than
0.001). Clinical manifestations of the disorder included depressed left
ventricular systolic function in five patients, ventricular arrhythmias in
five, systemic embolization in three, distinctive facial dysmorphism in
three, and familial recurrence in four patients. We conclude that isolated
noncompaction of left ventricular myocardium is a rare if not unique
disorder with characteristic morphological features that can be identified
by two-dimensional echocardiography. The incidence of cardiovascular
complications is high. The disorder may be associated with facial
dysmorphism and familial recurrence.
ARTICLES
Isolated noncompaction of left ventricular myocardium. A study of eight cases
Department of Pediatrics, UCLA Center for the Health Sciences.
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