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Randomized Controlled Trial
. 2022 Aug;77(8):805-811.
doi: 10.1136/thoraxjnl-2021-218196. Epub 2022 Mar 2.

Routine lung volume recruitment in boys with Duchenne muscular dystrophy: a randomised clinical trial

Sherri L Katz  1   2   3 , Jean K Mah  4   5   6 , Hugh J McMillan  7   2   3 , Craig Campbell  8   9 , Vid Bijelić  2 , Nick Barrowman  2   3 , Franco Momoli  3 , Henrietta Blinder  2 , Shawn D Aaron  3   10   11 , Laura C McAdam  12   13 , The Thanh Diem Nguyen  14 , Mark Tarnopolsky  15 , David F Wensley  16   17 , David Zielinski  18   19 , Louise Rose  20   21 , Nicole Sheers  22   23   24 , David J Berlowitz  25   26 , Lisa Wolfe  27   28 , Doug McKim  3   10   29
Affiliations
Randomized Controlled Trial

Routine lung volume recruitment in boys with Duchenne muscular dystrophy: a randomised clinical trial

Sherri L Katz et al. Thorax. 2022 Aug.

Abstract

Background: Impaired cough results in airway secretion retention, atelectasis and pneumonia in individuals with Duchenne muscular dystrophy (DMD). Lung volume recruitment (LVR) stacks breaths to inflate the lungs to greater volumes than spontaneous effort. LVR is recommended in DMD clinical care guidelines but is not well studied. We aimed to determine whether twice-daily LVR, compared with standard of care alone, attenuates the decline in FVC at 2 years in boys with DMD.

Methods: In this multicentre, assessor-blinded, randomised controlled trial, boys with DMD, aged 6-16 years with FVC >30% predicted, were randomised to receive conventional treatment or conventional treatment plus manual LVR twice daily for 2 years. The primary outcome was FVC % predicted at 2 years, adjusted for baseline FVC % predicted, age and ambulatory status. Secondary outcomes included change in chest wall distensibility (maximal insufflation capacity minus FVC) and peak cough flow.

Results: Sixty-six boys (36 in LVR group, 30 in control) were evaluated (median age (IQR): 11.5 years (9.5-13.5), median baseline FVC (IQR): 85% predicted (73-96)). Adjusted mean difference in FVC between groups at 2 years was 1.9% predicted (95% CI -6.9% to 10.7%; p=0.68) in the direction of treatment benefit. We found no differences in secondary outcomes.

Conclusion: There was no difference in decline in FVC % predicted with use of twice-daily LVR for boys with DMD and relatively normal lung function. The burden associated with routine LVR may outweigh the benefit. Benefits of LVR to maintain lung health in boys with worse baseline lung function still need to be clarified.

Trial registration number: NCT01999075.

Keywords: Duchenne muscular dystrophy; child; lung volume recruitment; randomized controlled trial; respiratory therapy.

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Conflict of interest statement

Competing interests: None declared.

Figures

Figure 1
Figure 1
CONSORT flow diagram. CONSORT, Consolidated Standards of Reporting Trials; LVR, lung volume recruitment; PFT, pulmonary function test.
Figure 2
Figure 2
Secondary analysis of FVC % predicted over time. Marginal means by group with 95% CI from mixed effect model of FVC % predicted, adjusted for age and ambulatory status, at 6-month intervals. LVR, lung volume recruitment.
Figure 3
Figure 3
Kaplan-Meier curve for a single missing data imputation. LVR, lung volume recruitment.
Figure 4
Figure 4
Change in secondary outcomes (MIC−VC (L), PCF-assisted – PCF-unassisted (L/min), MIP (cm H2O), MEP (cm H2O) and TLC (L) over time. Marginal means by group with 95% CI from mixed effect model of secondary outcomes, adjusted for age and ambulatory status, at 6-month intervals. MEP, maximal expiratory pressure; MIC, maximum insufflation capacity; MIP, maximal inspiratory pressure; PCF, peak cough flow; TLC, total lung capacity.
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References

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