Speckle-Tracking Echocardiography in Children With Duchenne Muscular Dystrophy: A Prospective Multicenter Controlled Cross-Sectional Study.


Journal

Journal of the American Society of Echocardiography : official publication of the American Society of Echocardiography
ISSN: 1097-6795
Titre abrégé: J Am Soc Echocardiogr
Pays: United States
ID NLM: 8801388

Informations de publication

Date de publication:
03 2019
Historique:
received: 01 07 2018
pubmed: 27 1 2019
medline: 14 8 2020
entrez: 26 1 2019
Statut: ppublish

Résumé

Prognosis of Duchenne muscular dystrophy (DMD) is related to cardiac dysfunction. Speckle-tracking echocardiographic (STE) imaging is emerging as a noninvasive functional biomarker to consider in the early detection of DMD-related cardiomyopathy. However, STE analysis has not been assessed in a prospectively controlled study, especially in presymptomatic children with DMD, and no study has used STE analysis in all three displacements (longitudinal, radial, and circumferential) and for both ventricles. This prospective controlled study enrolled 108 boys, 36 of whom had DMD (mean age, 11 ± 3.8 years) and 72 of whom were age-matched control subjects in a 1:2 case-control design. Conventional echocardiographic variables were collected for the left and right ventricles. STE analyses were performed in the longitudinal, radial, and circumferential displacements for the left ventricle and in the free wall longitudinal displacement for the right ventricle. The effect of age on the evolution of two-dimensional strain in children with DMD was studied by adding an interaction term, DMD × age, in the models. Conventional echocardiographic measures were normal in both groups. Left ventricular (LV) ejection fraction ranged from 45% to 76% (mean, 63 ± 6%) in the DMD group and from 55% to 76% (mean, 64 ± 5%) in the control group. Global LV strain mean measures were significantly worse in the DMD group for the longitudinal (-16.8 ± 3.9% vs -20.6 ± 2.6%, P < .0001), radial (22.7 ± 11.3% vs 31.7 ± 14%, P = .002), and circumferential (-16.5 ± 3.8% vs -20.3 ± 3.1%, P < .0001) displacements. The decrease of global LV longitudinal strain with age in children with DMD was 0.34% per year more marked than that in control subjects. The LV inferolateral and anterolateral segments were specifically impaired, especially in the basal area. Right ventricular function evaluated using conventional echocardiography and STE analysis was normal and not different between children with DMD and control subjects. The existence of altered LV strain despite normal LV function in children with DMD represents an important perspective for future pediatric drug trials in DMD-related cardiomyopathy prevention.

Sections du résumé

BACKGROUND
Prognosis of Duchenne muscular dystrophy (DMD) is related to cardiac dysfunction. Speckle-tracking echocardiographic (STE) imaging is emerging as a noninvasive functional biomarker to consider in the early detection of DMD-related cardiomyopathy. However, STE analysis has not been assessed in a prospectively controlled study, especially in presymptomatic children with DMD, and no study has used STE analysis in all three displacements (longitudinal, radial, and circumferential) and for both ventricles.
METHODS
This prospective controlled study enrolled 108 boys, 36 of whom had DMD (mean age, 11 ± 3.8 years) and 72 of whom were age-matched control subjects in a 1:2 case-control design. Conventional echocardiographic variables were collected for the left and right ventricles. STE analyses were performed in the longitudinal, radial, and circumferential displacements for the left ventricle and in the free wall longitudinal displacement for the right ventricle. The effect of age on the evolution of two-dimensional strain in children with DMD was studied by adding an interaction term, DMD × age, in the models.
RESULTS
Conventional echocardiographic measures were normal in both groups. Left ventricular (LV) ejection fraction ranged from 45% to 76% (mean, 63 ± 6%) in the DMD group and from 55% to 76% (mean, 64 ± 5%) in the control group. Global LV strain mean measures were significantly worse in the DMD group for the longitudinal (-16.8 ± 3.9% vs -20.6 ± 2.6%, P < .0001), radial (22.7 ± 11.3% vs 31.7 ± 14%, P = .002), and circumferential (-16.5 ± 3.8% vs -20.3 ± 3.1%, P < .0001) displacements. The decrease of global LV longitudinal strain with age in children with DMD was 0.34% per year more marked than that in control subjects. The LV inferolateral and anterolateral segments were specifically impaired, especially in the basal area. Right ventricular function evaluated using conventional echocardiography and STE analysis was normal and not different between children with DMD and control subjects.
CONCLUSIONS
The existence of altered LV strain despite normal LV function in children with DMD represents an important perspective for future pediatric drug trials in DMD-related cardiomyopathy prevention.

Identifiants

pubmed: 30679141
pii: S0894-7317(18)30608-4
doi: 10.1016/j.echo.2018.10.017
pii:
doi:

Types de publication

Controlled Clinical Trial Journal Article Multicenter Study Research Support, Non-U.S. Gov't

Langues

eng

Sous-ensembles de citation

IM

Pagination

412-422

Informations de copyright

Copyright © 2018 American Society of Echocardiography. Published by Elsevier Inc. All rights reserved.

Auteurs

Pascal Amedro (P)

Pediatric and Congenital Cardiology Department, M3C Regional Reference CHD Center, CHU Montpellier, Montpellier, France; PHYMEDEXP, University of Montpellier, CNRS, INSERM, CHU Montpellier, Montpellier, France. Electronic address: p-amedro@chu-montpellier.fr.

Marie Vincenti (M)

Pediatric and Congenital Cardiology Department, M3C Regional Reference CHD Center, CHU Montpellier, Montpellier, France; PHYMEDEXP, University of Montpellier, CNRS, INSERM, CHU Montpellier, Montpellier, France; Pediatric Cardiology and Rehabilitation Unit, St-Pierre Institute, Palavas-Les-Flots, France.

Gregoire De La Villeon (G)

Pediatric and Congenital Cardiology Department, M3C Regional Reference CHD Center, CHU Montpellier, Montpellier, France; Pediatric Cardiology and Rehabilitation Unit, St-Pierre Institute, Palavas-Les-Flots, France.

Kathleen Lavastre (K)

Pediatric and Congenital Cardiology Department, M3C Regional Reference CHD Center, CHU Montpellier, Montpellier, France.

Catherine Barrea (C)

Pediatric and Congenital Cardiology Department, Cliniques Universitaires Saint-Luc, UCL University, Brussels, Belgium.

Sophie Guillaumont (S)

Pediatric and Congenital Cardiology Department, M3C Regional Reference CHD Center, CHU Montpellier, Montpellier, France; Pediatric Cardiology and Rehabilitation Unit, St-Pierre Institute, Palavas-Les-Flots, France.

Charlene Bredy (C)

Pediatric and Congenital Cardiology Department, M3C Regional Reference CHD Center, CHU Montpellier, Montpellier, France; Department of Cardiology, CHU Montpellier, Montpellier, France.

Lucie Gamon (L)

Epidemiology and Clinical Research Department, CHU Montpellier, Montpellier, France.

Albano C Meli (AC)

PHYMEDEXP, University of Montpellier, CNRS, INSERM, CHU Montpellier, Montpellier, France.

Olivier Cazorla (O)

PHYMEDEXP, University of Montpellier, CNRS, INSERM, CHU Montpellier, Montpellier, France.

Jeremy Fauconnier (J)

PHYMEDEXP, University of Montpellier, CNRS, INSERM, CHU Montpellier, Montpellier, France.

Pierre Meyer (P)

PHYMEDEXP, University of Montpellier, CNRS, INSERM, CHU Montpellier, Montpellier, France; Pediatric Neurology, National Reference Centre for Neuromuscular Diseases, CHU Montpellier, Montpellier, France.

François Rivier (F)

PHYMEDEXP, University of Montpellier, CNRS, INSERM, CHU Montpellier, Montpellier, France; Pediatric Neurology, National Reference Centre for Neuromuscular Diseases, CHU Montpellier, Montpellier, France.

Jerome Adda (J)

Department of Cardiology, CHU Montpellier, Montpellier, France.

Thibault Mura (T)

Epidemiology and Clinical Research Department, CHU Montpellier, Montpellier, France; Clinical Investigation Center, University of Montpellier, INSERM, CHU Montpellier, Montpellier, France.

Alain Lacampagne (A)

PHYMEDEXP, University of Montpellier, CNRS, INSERM, CHU Montpellier, Montpellier, France.

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Classifications MeSH