Association between age at loss of ambulation and cardiac function in adults with Duchenne muscular dystrophy

Cardiomyopathy is a common co-morbidity in individuals with Duchenne muscular dystrophy (DMD). This retrospective single centre study investigated the relationship between age at loss of ambulation (LOA) and late stage left ventricular ejection fraction (LVEF) in 84 individuals (> 16 years old) with DMD taking glucocorticoid and ACE inhibitors treatment. Regression analyses showed a positive correlation between later age at LOA and higher LVEF in adulthood (linear regression estimate 1.49, 95 % CI: 0.13-2.84, p = 0.03). Each additional year of ambulation increased the odds of displaying a higher LVEF category (LVEF 40 %, 40 - 50 % or 50 %) by 35 % (p = 0.003). Sensitivity models excluding cardioprotective genotypes (absence of Dp116 isoform) and mild motor phenotypes (out of frame deletions amenable to skip exon 44 and 45) confirmed this association while models including age at respiratory impairment did not improve the model. Individuals who lost ambulation before age 11.92 (ROC AUC 0.73, 95 % CI: 0.60-0.85) reached a LVEF <40 % 5.21 years earlier than those who lost ambulation after that age (adjusted restricted mean survival time 19.08 vs 24.29 years, p < 0.001). These findings may suggest that prolonging ambulation does not impact cardiac function adversely in advance stages of DMD.

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Publication Details

Journal
Neuromuscular Disorders
Published
2025-01-01
DOI
https://doi.org/10.1016/j.nmd.2025.105276
Citations
2
Primary Topic
Muscle Physiology and Disorders
Type
article
Field-Weighted Citation Impact
1.61

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article

Association between age at loss of ambulation and cardiac function in adults with Duchenne muscular dystrophy

John Bourke, Michela Guglieri, Volker Straub, Kadhim Kadhim et al.
2 citations
Neuromuscular Disorders
Muscle Physiology and Disorders
1.61
article

Association between age at loss of ambulation and cardiac function in adults with Duchenne muscular dystrophy

John Bourke, Michela Guglieri, Volker Straub, Kadhim Kadhim, Giorgio Tasca, Jordi Díaz‐Manera, Anna Johnson, C. Marini Bettolo, Maha A. Elseed, Marianela Schiava
article en
2 citations

Abstract

Cardiomyopathy is a common co-morbidity in individuals with Duchenne muscular dystrophy (DMD). This retrospective single centre study investigated the relationship between age at loss of ambulation (LOA) and late stage left ventricular ejection fraction (LVEF) in 84 individuals (> 16 years old) with DMD taking glucocorticoid and ACE inhibitors treatment. Regression analyses showed a positive correlation between later age at LOA and higher LVEF in adulthood (linear regression estimate 1.49, 95 % CI: 0.13-2.84, p = 0.03). Each additional year of ambulation increased the odds of displaying a higher LVEF category (LVEF 40 %, 40 - 50 % or 50 %) by 35 % (p = 0.003). Sensitivity models excluding cardioprotective genotypes (absence of Dp116 isoform) and mild motor phenotypes (out of frame deletions amenable to skip exon 44 and 45) confirmed this association while models including age at respiratory impairment did not improve the model. Individuals who lost ambulation before age 11.92 (ROC AUC 0.73, 95 % CI: 0.60-0.85) reached a LVEF <40 % 5.21 years earlier than those who lost ambulation after that age (adjusted restricted mean survival time 19.08 vs 24.29 years, p < 0.001). These findings may suggest that prolonging ambulation does not impact cardiac function adversely in advance stages of DMD.

Neuromuscular DisordersVol. 46
Newcastle upon Tyne Hospitals NHS Foundation Trust (GB), NIHR Newcastle Biomedical Research Centre (GB)
Duchenne Research Fund, University College London
Openalex Percentile: Top 20%
Muscle Physiology and Disorders
1.61
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