---
title: "Improved survival in Duchenne muscular dystrophy: meta-analysis of eras and impact of home ventila"
id: "pubmed-42701154"
canonical_url: "https://medichelpline.com/clinical-feed/pubmed-42701154"
content_type: "clinical_feed_article"
specialty: "Pharmacology"
source_name: "PubMed / NCBI"
source_url: "https://pubmed.ncbi.nlm.nih.gov/42701154/"
doi: "10.1007/s00415-026-14121-4"
published_at: "2026-09-06T00:00:00.000Z"
evidence_level: "Journal Article"
license: "CC-BY-NC-4.0 / Informational Use"
---
# Improved survival in Duchenne muscular dystrophy: meta-analysis of eras and impact of home ventila
## Provenance & Clinical Metadata
- **Canonical URL:** https://medichelpline.com/clinical-feed/pubmed-42701154
- **Specialty:** [Pharmacology](https://medichelpline.com/clinical-feed/pharmacology.md)
- **Primary Source:** PubMed / NCBI
- **Source URL:** [Original Journal Publication](https://pubmed.ncbi.nlm.nih.gov/42701154/)
- **DOI:** [10.1007/s00415-026-14121-4](https://doi.org/10.1007%2Fs00415-026-14121-4)
- **Published At:** 2026-09-06T00:00:00.000Z
- **Evidence Rating:** Journal Article
## Executive GIST (TL;DR)
- This systematic review and cumulative meta-analysis examined temporal changes in survival for patients with **Duchenne muscular dystrophy (DMD)** and the impact of **home mechanical ventilation (HMV)**. - The review searched PubMed from 1977 through 13 October 2025 and was registered on PROSPERO (CRD420251163011). - Fifty-three studies were included, with a median follow-up of 8 years and encompassing more than 13,000 patients; about 60% of patients received HMV. - Pooled median survival differed markedly by ventilation status: ventilated patients had a pooled median survival of 29 years (95% CI 27–31) versus 19 years (95% CI 18–20) for non-ventilated patients. - Survival improved over time in both ventilated and non-ventilated groups, indicating progressive gains across treatment eras. - **Glucocorticoid** therapy was not associated with improved survival in the analyses reported (p = 0.45). - Use of cardiac therapies—specifically **renin-angiotensin system inhibitors** (p = 0.002) and **β-blockers** (p = 0.02)—was associated with longer survival. - The leading cause of death shifted from respiratory failure toward cardiac causes as respiratory care improved; enhanced cardiac management corresponded with a larger share of other non-respiratory causes. - The authors conclude that median survival now approaches the third decade in ventilated patients and emphasize the need for long-term, multidisciplinary care with early cardioprotective strategies. - Study-level risk of bias was assessed using the Newcastle-Ottawa Scale. Specific methodological details beyond the abstract (e.g., selection criteria, heterogeneity metrics, sensitivity analyses) were not reported in the provided source text.
## Clinical Analysis & Structured Key Points
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Affiliations Expand ### Affiliations * 1 Sleep Disorders Centre, Department of Pulmonology, University Hospital Zurich, Raemistrasse 100, 8091, Zurich, Switzerland. * 2 University of Zurich, Raemistrasse 71, 8006, Zurich, Switzerland. * 3 Sleep Disorders Centre, Department of Pulmonology, University Hospital Zurich, Raemistrasse 100, 8091, Zurich, Switzerland. estherirene.schwarz@usz.ch. * 4 University of Zurich, Raemistrasse 71, 8006, Zurich, Switzerland. estherirene.schwarz@usz.ch. * PMID: **42701154** * DOI: [ 10.1007/s00415-026-14121-4 ](https://doi.org/10.1007/s00415-026-14121-4) Item in Clipboard Meta-Analysis # Improving survival in Duchenne muscular dystrophy across eras: a systematic review and cumulative meta-analysis Lara Benning et al. J Neurol. 2026. Show details Display options Display options Format Abstract PubMed PMID J Neurol Actions * [ Search in PubMed ](https://pubmed.ncbi.nlm.nih.gov/?term=%22J+Neurol%22%5Bjour%5D&sort=date&sort_order=desc) * [ Search in NLM Catalog ](https://www.ncbi.nlm.nih.gov/nlmcatalog?term=%22J+Neurol%22%5BTitle+Abbreviation%5D) * [ Add to Search ](https://pubmed.ncbi.nlm.nih.gov/42701154/) . 2026 Sep 6;273(10):564. doi: 10.1007/s00415-026-14121-4. ### Authors [Lara Benning](https://pubmed.ncbi.nlm.nih.gov/?term=Benning+L&cauthor_id=42701154)[ 1 ](https://pubmed.ncbi.nlm.nih.gov/42701154/#short-view-affiliation-1 "Sleep Disorders Centre, Department of Pulmonology, University Hospital Zurich, Raemistrasse 100, 8091, Zurich, Switzerland.")[ 2 ](https://pubmed.ncbi.nlm.nih.gov/42701154/#short-view-affiliation-2 "University of Zurich, Raemistrasse 71, 8006, Zurich, Switzerland."), [Zoe Bousraou](https://pubmed.ncbi.nlm.nih.gov/?term=Bousraou+Z&cauthor_id=42701154)[ 1 ](https://pubmed.ncbi.nlm.nih.gov/42701154/#short-view-affiliation-1 "Sleep Disorders Centre, Department of Pulmonology, University Hospital Zurich, Raemistrasse 100, 8091, Zurich, Switzerland.")[ 2 ](https://pubmed.ncbi.nlm.nih.gov/42701154/#short-view-affiliation-2 "University of Zurich, Raemistrasse 71, 8006, Zurich, Switzerland."), [Bastian Gruber](https://pubmed.ncbi.nlm.nih.gov/?term=Gruber+B&cauthor_id=42701154)[ 1 ](https://pubmed.ncbi.nlm.nih.gov/42701154/#short-view-affiliation-1 "Sleep Disorders Centre, Department of Pulmonology, University Hospital Zurich, Raemistrasse 100, 8091, Zurich, Switzerland.")[ 2 ](https://pubmed.ncbi.nlm.nih.gov/42701154/#short-view-affiliation-2 "University of Zurich, Raemistrasse 71, 8006, Zurich, Switzerland."), [Silvia Ulrich](https://pubmed.ncbi.nlm.nih.gov/?term=Ulrich+S&cauthor_id=42701154)[ 1 ](https://pubmed.ncbi.nlm.nih.gov/42701154/#short-view-affiliation-1 "Sleep Disorders Centre, Department of Pulmonology, University Hospital Zurich, Raemistrasse 100, 8091, Zurich, Switzerland.")[ 2 ](https://pubmed.ncbi.nlm.nih.gov/42701154/#short-view-affiliation-2 "University of Zurich, Raemistrasse 71, 8006, Zurich, Switzerland."), [Esther I Schwarz](https://pubmed.ncbi.nlm.nih.gov/?term=Schwarz+EI&cauthor_id=42701154)[ 3 ](https://pubmed.ncbi.nlm.nih.gov/42701154/#short-view-affiliation-3 "Sleep Disorders Centre, Department of Pulmonology, University Hospital Zurich, Raemistrasse 100, 8091, Zurich, Switzerland. estherirene.schwarz@usz.ch.")[ 4 ](https://pubmed.ncbi.nlm.nih.gov/42701154/#short-view-affiliation-4 "University of Zurich, Raemistrasse 71, 8006, Zurich, Switzerland. estherirene.schwarz@usz.ch.") ### Affiliations * 1 Sleep Disorders Centre, Department of Pulmonology, University Hospital Zurich, Raemistrasse 100, 8091, Zurich, Switzerland. * 2 University of Zurich, Raemistrasse 71, 8006, Zurich, Switzerland. * 3 Sleep Disorders Centre, Department of Pulmonology, University Hospital Zurich, Raemistrasse 100, 8091, Zurich, Switzerland. estherirene.schwarz@usz.ch. * 4 University of Zurich, Raemistrasse 71, 8006, Zurich, Switzerland. estherirene.schwarz@usz.ch. * PMID: **42701154** * DOI: [ 10.1007/s00415-026-14121-4 ](https://doi.org/10.1007/s00415-026-14121-4) Item in Clipboard Cite Display options Display options Format Abstract PubMed PMID ## Abstract **Background:** Duchenne muscular dystrophy (DMD) was historically associated with death in the late teens or early twenties, mainly from respiratory failure. Survival has improved substantially with home mechanical ventilation (HMV) and multidisciplinary care, although variability remains. This study evaluated temporal trends in survival in DMD and the impact of HMV. **Methods:** A study-level cumulative meta-analysis (PROSPERO CRD420251163011) of studies reporting survival outcomes in patients with DMD was conducted (PubMed 1977 to 13 October 2025). Pooled estimates of median survival were calculated, and random-effects meta-analyses with predefined subgroups (HMV and study period) were performed, alongside meta-regressions. Risk of bias was assessed using the Newcastle-Ottawa Scale. **Results:** 53 studies (median follow-up 8 years), comprising more than 13,000 patients, of whom 60% received HMV, were included. Median survival differed substantially between ventilated (29 years, 95%CI 27 to 31) and non-ventilated (19 years, 95%CI 18 to 20) patients. Survival improved progressively over time in both groups. Glucocorticoid therapy was not associated with improved survival (p=0.45), whereas treatment with heart failure medications, including renin-angiotensin system inhibitors (p=0.002) and β-blockers (p=0.02), was associated with longer survival. The predominance of mortality shifted from respiratory to cardiac causes, while enhanced cardiac management was associated with a growing contribution of other causes of death. **Conclusion:** Survival in DMD has increased substantially over time, with median survival now approaching the third decade of life among ventilated patients. The growing contribution of cardiac and other non-respiratory causes of death highlights the importance of long-term multidisciplinary and early cardioprotective intervention. **Study registration:** The meta-analysis and systematic review have been registered on PROSPERO (CRD420251163011). **Keywords:** Cardiomyopathy; Duchenne muscular dystrophy; Home mechanical ventilation; Life expectancy; Mortality; Survival. © 2026. The Author(s). [PubMed Disclaimer](https://pubmed.ncbi.nlm.nih.gov/disclaimer/) ## Conflict of interest statement Declarations. Conflicts of interest: The authors declare that they have no conflict of interest. Ethical standards: Included studies have been approved by the appropriate ethics committee and have therefore been performed in accordance with the ethical standards laid down in the 1964 Declaration of Helsinki and its later amendments. ## References 1. 1. Hoffman EP, Brown RH Jr., Kunkel LM (1987) Dystrophin: the protein product of the Duchenne muscular dystrophy locus. Cell 51:919–928. - [DOI](https://doi.org/10.1016/0092-8674\(87\)90579-4) - [PubMed](https://pubmed.ncbi.nlm.nih.gov/3319190/) 2. 1. Duan D, Goemans N, Takeda S, Mercuri E, Aartsma-Rus A (2021) Duchenne muscular dystrophy. Nat Rev Dis Primers 7:13. - [DOI](https://doi.org/10.1038/s41572-021-00248-3) - [PubMed](https://pubmed.ncbi.nlm.nih.gov/33602943/) - [PMC](https://pmc.ncbi.nlm.nih.gov/articles/10557455/) 3. 1. Eagle M, Baudouin SV, Chandler C, Giddings DR, Bullock R, Bushby K (1967) Survival in Duchenne muscular dystrophy: improvements in life expectancy since 1967 and the impact of home nocturnal ventilation. Neuromuscul Disord 12:926–929. - [DOI](https://doi.org/10.1016/s0960-8966\(02\)00140-2) 4. 1. Vianello A, Bevilacqua M, Salvador V, Cardaioli C, Vincenti E (1994) Long-term nasal intermittent positive pressure ventilation in advanced Duchenne’s muscular dystrophy. Chest 105:445–448. - [DOI](https://doi.org/10.1378/chest.105.2.445) - [PubMed](https://pubmed.ncbi.nlm.nih.gov/8306744/) 5. 1. Simonds AK, Muntoni F, Heather S, Fielding S (1998) Impact of nasal ventilation on survival in hypercapnic Duchenne muscular dystrophy. Thorax 53:949–952. - [DOI](https://doi.org/10.1136/thx.53.11.949) - [PubMed](https://pubmed.ncbi.nlm.nih.gov/10193393/) - [PMC](https://pmc.ncbi.nlm.nih.gov/articles/1745110/) Show all 79 references ## Publication types * Systematic Review Actions * [ Search in PubMed ](https://pubmed.ncbi.nlm.nih.gov/?term=%22Systematic+Review%22%5Bpt%5D&sort=date&sort_order=desc) * [ Search in MeSH ](https://www.ncbi.nlm.nih.gov/mesh?term=Systematic+Review) * [ Add to Search ](https://pubmed.ncbi.nlm.nih.gov/42701154/) * Meta-Analysis Actions * [ Search in PubMed ](https://pubmed.ncbi.nlm.nih.gov/?term=%22Meta-Analysis%22%5Bpt%5D&sort=date&sort_order=desc) * [ Search in MeSH ](https://www.ncbi.nlm.nih.gov/mesh?term=Meta-Analysis) * [ Add to Search ](https://pubmed.ncbi.nlm.nih.gov/42701154/) ## MeSH terms * Humans Actions * [ Search in PubMed ](https://pubmed.ncbi.nlm.nih.gov/?term=%22Humans%22%5BMeSH%5D&sort=date&sort_order=desc) * [ Search in MeSH ](https://www.ncbi.nlm.nih.gov/mesh?term=Humans) * [ Add to Search ](https://pubmed.ncbi.nlm.nih.gov/42701154/) * Muscular Dystrophy, Duchenne* / complications Actions * [ Search in PubMed ](https://pubmed.ncbi.nlm.nih.gov/?term=%22Muscular+Dystrophy%2C+Duchenne%2Fcomplications%22%5BMAJR%5D&sort=date&sort_order=desc) * [ Search in MeSH ](https://www.ncbi.nlm.nih.gov/mesh?term=Muscular+Dystrophy%2C+Duchenne) * [ Add to Search ](https://pubmed.ncbi.nlm.nih.gov/42701154/) * Muscular Dystrophy, Duchenne* / mortality Actions * [ Search in PubMed ](https://pubmed.ncbi.nlm.nih.gov/?term=%22Muscular+Dystrophy%2C+Duchenne%2Fmortality%22%5BMAJR%5D&sort=date&sort_order=desc) * [ Search in MeSH ](https://www.ncbi.nlm.nih.gov/mesh?term=Muscular+Dystrophy%2C+Duchenne) * [ Add to Search ](https://pubmed.ncbi.nlm.nih.gov/42701154/) * Muscular Dystrophy, Duchenne* / therapy Actions * [ Search in PubMed ](https://pubmed.ncbi.nlm.nih.gov/?term=%22Muscular+Dystrophy%2C+Duchenne%2Ftherapy%22%5BMAJR%5D&sort=date&sort_order=desc) * [ Search in MeSH ](https://www.ncbi.nlm.nih.gov/mesh?term=Muscular+Dystrophy%2C+Duchenne) * [ Add to Search ](https://pubmed.ncbi.nlm.nih.gov/42701154/) * Respiration, Artificial* Actions * [ Search in PubMed ](https://pubmed.ncbi.nlm.nih.gov/?term=%22Respiration%2C+Artificial%22%5BMAJR%5D&sort=date&sort_order=desc) * [ Search in MeSH ](https://www.ncbi.nlm.nih.gov/mesh?term=Respiration%2C+Artificial) * [ Add to Search ](https://pubmed.ncbi.nlm.nih.gov/42701154/) * Respiratory Insufficiency* / etiology Actions * [ Search in PubMed ](https://pubmed.ncbi.nlm.nih.gov/?term=%22Respiratory+Insufficiency%2Fetiology%22%5BMAJR%5D&sort=date&sort_order=desc) * [ Search in MeSH ](https://www.ncbi.nlm.nih.gov/mesh?term=Respiratory+Insufficiency) * [ Add to Search ](https://pubmed.ncbi.nlm.nih.gov/42701154/) * Respiratory Insufficiency* / mortality Actions * [ Search in PubMed ](https://pubmed.ncbi.nlm.nih.gov/?term=%22Respiratory+Insufficiency%2Fmortality%22%5BMAJR%5D&sort=date&sort_order=desc) * [ Search in MeSH ](https://www.ncbi.nlm.nih.gov/mesh?term=Respiratory+Insufficiency) * [ Add to Search ](https://pubmed.ncbi.nlm.nih.gov/42701154/) * Respiratory Insufficiency* / therapy Actions * [ Search in PubMed ](https://pubmed.ncbi.nlm.nih.gov/?term=%22Respiratory+Insufficiency%2Fthe
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