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

Sherri L. Katz; Jean K. Mah; Hugh J. McMillan; Craig Campbell; Vid Bijelić; Nick Barrowman; Franco Momoli; Henrietta Blinder; Shawn D. Aaron; Laura C. McAdam; The Thanh Diem Nguyen; Mark Tarnopolsky; David F. Wensley; David Zielinski; Louise Rose; Nicole Sheers; David J. Berlowitz; Lisa Wolfe; Doug McKim

doi:10.1136/thoraxjnl-2021-218196

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

Sherri L. Katz^*, Jean K. Mah, Hugh J. McMillan, Craig Campbell, Vid Bijelić, Nick Barrowman, Franco Momoli, Henrietta Blinder, Shawn D. Aaron, Laura C. McAdam, The Thanh Diem Nguyen, Mark Tarnopolsky, David F. Wensley, David Zielinski, Louise Rose, Nicole Sheers, David J. Berlowitz, Lisa Wolfe, Doug McKim

^*Corresponding author for this work

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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.

Original language	English
Pages (from-to)	805-811
Number of pages	7
Journal	Thorax
Volume	77
Issue number	8
Early online date	25 Jul 2022
DOIs	https://doi.org/10.1136/thoraxjnl-2021-218196
Publication status	Published - 1 Aug 2022

Keywords

child
Duchenne muscular dystrophy
lung volume recruitment
randomized controlled trial
respiratory therapy

UN SDGs

This output contributes to the following UN Sustainable Development Goals (SDGs)

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Katz, S. L., Mah, J. K., McMillan, H. J., Campbell, C., Bijelić, V., Barrowman, N., Momoli, F., Blinder, H., Aaron, S. D., McAdam, L. C., Nguyen, T. T. D., Tarnopolsky, M., Wensley, D. F., Zielinski, D., Rose, L., Sheers, N., Berlowitz, D. J., Wolfe, L., & McKim, D. (2022). Routine lung volume recruitment in boys with Duchenne muscular dystrophy: a randomised clinical trial. Thorax, 77(8), 805-811. https://doi.org/10.1136/thoraxjnl-2021-218196

@article{509d034e0a6a4562a9fb50e83440f54a,

title = "Routine lung volume recruitment in boys with Duchenne muscular dystrophy: a randomised clinical trial",

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 = "child, Duchenne muscular dystrophy, lung volume recruitment, randomized controlled trial, respiratory therapy",

author = "Katz, {Sherri L.} and Mah, {Jean K.} and McMillan, {Hugh J.} and Craig Campbell and Vid Bijeli{\'c} and Nick Barrowman and Franco Momoli and Henrietta Blinder and Aaron, {Shawn D.} and McAdam, {Laura C.} and Nguyen, {The Thanh Diem} and Mark Tarnopolsky and Wensley, {David F.} and David Zielinski and Louise Rose and Nicole Sheers and Berlowitz, {David J.} and Lisa Wolfe and Doug McKim",

year = "2022",

month = aug,

day = "1",

doi = "10.1136/thoraxjnl-2021-218196",

language = "English",

volume = "77",

pages = "805--811",

journal = "Thorax",

issn = "0040-6376",

publisher = "BRITISH MED JOURNAL PUBL GROUP",

number = "8",

}

Katz, SL, Mah, JK, McMillan, HJ, Campbell, C, Bijelić, V, Barrowman, N, Momoli, F, Blinder, H, Aaron, SD, McAdam, LC, Nguyen, TTD, Tarnopolsky, M, Wensley, DF, Zielinski, D, Rose, L, Sheers, N, Berlowitz, DJ, Wolfe, L & McKim, D 2022, 'Routine lung volume recruitment in boys with Duchenne muscular dystrophy: a randomised clinical trial', Thorax, vol. 77, no. 8, pp. 805-811. https://doi.org/10.1136/thoraxjnl-2021-218196

TY - JOUR

T1 - Routine lung volume recruitment in boys with Duchenne muscular dystrophy

T2 - a randomised clinical trial

AU - Katz, Sherri L.

AU - Mah, Jean K.

AU - McMillan, Hugh J.

AU - Campbell, Craig

AU - Bijelić, Vid

AU - Barrowman, Nick

AU - Momoli, Franco

AU - Blinder, Henrietta

AU - Aaron, Shawn D.

AU - McAdam, Laura C.

AU - Nguyen, The Thanh Diem

AU - Tarnopolsky, Mark

AU - Wensley, David F.

AU - Zielinski, David

AU - Rose, Louise

AU - Sheers, Nicole

AU - Berlowitz, David J.

AU - Wolfe, Lisa

AU - McKim, Doug

PY - 2022/8/1

Y1 - 2022/8/1

N2 - 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.

AB - 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.

KW - child

KW - Duchenne muscular dystrophy

KW - lung volume recruitment

KW - randomized controlled trial

KW - respiratory therapy

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U2 - 10.1136/thoraxjnl-2021-218196

DO - 10.1136/thoraxjnl-2021-218196

M3 - Article

C2 - 35236763

AN - SCOPUS:85130997156

SN - 0040-6376

VL - 77

SP - 805

EP - 811

JO - Thorax

JF - Thorax

IS - 8

ER -

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

Abstract

Keywords

UN SDGs

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