Trial reportPediatric pulmonology2017
Adherence and barriers to hyperinsufflation in children with congenital muscular dystrophy.
Trial report in Pediatric pulmonology, 2017. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 3 papers.
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The abstract states no effect estimate the extractor could read, or names no intervention and outcome on the map, so this paper lights no cell and moves no belief. It is still indexed, cited and linked below.
The trial behind it
Trials whose registry record cites this paper, or whose number appears in the abstract. A trial that started after this paper was published is citing it as background, not reporting it.
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Who cites it
3 citing papers in PubMed.
- Patterns of Adherence to Lung Volume Recruitment Therapy Amongst Individuals With Duchenne Muscular Dystrophy: A Secondary Analysis of the STEADFAST Randomized Controlled Trial.Pediatric pulmonology · 2025Trial
- Randomized trial of lung hyperinflation therapy in children with congenital muscular dystrophy.Pediatric pulmonology · 2020Trial
- Improving Nocturnal Non-Invasive Positive Pressure Support Adherence in Children and Young Adults With Duchenne Muscular Dystrophy.Pediatric pulmonology · 2026Article
Corrections and comments
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Authors and funding
9 authors.
Funding
Abstract
backgroundCongenital muscular dystrophy (CMD) is a rare, inherited neuromuscular disease characterized by progressive muscle weakness, thoracic insufficiency, and ultimately respiratory failure. Adherence to respiratory therapies in children with neuromuscular disorders is unknown. This study examined the multimodal assessment of adherence and barriers to 15 min, twice daily hyperinsufflation in children with CMD. Adherence was hypothesized to be greater than 50% and discomfort, embarrassment, and difficulty finding time were hypothesized to be barriers.
methodsParticipants included 18 children with CMD. Personalized hyperinsufflation settings were determined based on pressure-volume measurements at each study visit. Adherence was measured by a daily phone diary (DPD) and by electronic data download from the hyperinsufflation device. The DPD was conducted twice over a 48-hr period to capture a weekend and weekday, with the goal being 60 min of hyperinsufflation over the 48 hr (100% adherence). The hyperinsufflation objective electronic data reflected daily use of hyperinsufflation for the same 48-hr period. Data from DPD and the corresponding hyperinsufflation device data were used for analyses.
resultsAdherence to hyperinsufflation was 40% via DPD and 44% for electronic data, with strong convergence between methods (r = 0.75, P < 0.001). Surprisingly, 53% of participants reported no barriers despite low adherence. Social distractions and family obligations were identified as barriers. There were no differences in adherence between those who did and did not endorse barriers to hyperinsufflation (DPD: t(13) = 0.44, P = n.s.; hyperinsufflation device: t(13) = -0.23, P = n.s.).
conclusionAdherence to hyperinsufflation is a significant problem in children with CMD and families have difficulty identifying adherence barriers. An important next step is to encourage open dialog around adherence barriers and promote adherence behaviors via intervention. Pediatr Pulmonol. 2017; 52:939-945. © 2016 Wiley Periodicals, Inc.
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