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

Cough / etiology Humans Lung Volume Measurements Male Muscular Dystrophy, Duchenne / complications Respiratory Function Tests / methods Vital Capacity
Duchenne muscular dystrophy child lung volume recruitment randomized controlled trial respiratory therapy

Journal

Thorax
ISSN: 1468-3296
Titre abrégé: Thorax
Pays: England
ID NLM: 0417353

Informations de publication

Date de publication:
08 2022
Historique:
received: 08 09 2021
accepted: 25 01 2022
pubmed: 4 3 2022
medline: 28 7 2022
entrez: 3 3 2022
Statut: ppublish

Résumé

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. 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. 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. 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. NCT01999075.

Sections du résumé

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.

Identifiants

DOI: 10.1136/thoraxjnl-2021-218196 PMID: 35236763 PMC: PMC9340020
pubmed: 35236763
pii: thoraxjnl-2021-218196
doi: 10.1136/thoraxjnl-2021-218196
pmc: PMC9340020
doi:

Banques de données

ClinicalTrials.gov
['NCT01999075']

Types de publication

Journal Article Multicenter Study Randomized Controlled Trial Research Support, Non-U.S. Gov't

Langues

eng

Sous-ensembles de citation

IM

Pagination

805-811

Commentaires et corrections

Type : CommentIn

Informations de copyright

© Author(s) (or their employer(s)) 2022. Re-use permitted under CC BY-NC. No commercial re-use. See rights and permissions. Published by BMJ.

Déclaration de conflit d'intérêts

Competing interests: None declared.

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Auteurs

Sherri L Katz (SL)

Department of Pediatrics, Children's Hospital of Eastern Ontario, Ottawa, Ontario, Canada skatz@cheo.on.ca.
CHEO Research Institute, Ottawa, Ontario, Canada.
Faculty of Medicine, University of Ottawa, Ottawa, Ontario, Canada.

Jean K Mah (JK)

Division of Pediatric Neurology, Alberta Children's Hospital, Calgary, Alberta, Canada.
Department of Pediatric and Clinical Neurosciences, University of Calgary Cumming School of Medicine, Calgary, Alberta, Canada.
Alberta Children's Hospital Research Institute, Calgary, Alberta, Canada.

Hugh J McMillan (HJ)

Department of Pediatrics, Children's Hospital of Eastern Ontario, Ottawa, Ontario, Canada.
CHEO Research Institute, Ottawa, Ontario, Canada.
Faculty of Medicine, University of Ottawa, Ottawa, Ontario, Canada.

Craig Campbell (C)

Department of Pediatrics, Epidemiology and Clinical Neurological Sciences, University of Western Ontario, London, Ontario, Canada.
Department of Pediatrics, London Health Sciences Centre Children's Hospital, London, Ontario, Canada.

Vid Bijelić (V)

CHEO Research Institute, Ottawa, Ontario, Canada.

Nick Barrowman (N)

CHEO Research Institute, Ottawa, Ontario, Canada.
Faculty of Medicine, University of Ottawa, Ottawa, Ontario, Canada.

Franco Momoli (F)

Faculty of Medicine, University of Ottawa, Ottawa, Ontario, Canada.

Henrietta Blinder (H)

CHEO Research Institute, Ottawa, Ontario, Canada.
ORCID: 0000-0001-6307-6094

Shawn D Aaron (SD)

Faculty of Medicine, University of Ottawa, Ottawa, Ontario, Canada.
Ottawa Hospital Research Institute, Ottawa, Ontario, Canada.
Division of Respirology, Department of Medicine, The Ottawa Hospital, Ottawa, Ontario, Canada.

Laura C McAdam (LC)

Holland Bloorview Kids Rehabilitation Hospital, Toronto, Ontario, Canada.
Department of Pediatrics, University of Toronto, Toronto, Ontario, Canada.

The Thanh Diem Nguyen (TTD)

Department of Respiratory Medicine, Centre Hospitalier Universitaire Sainte-Justine, Montreal, Quebec, Canada.

Mark Tarnopolsky (M)

Division of Neuromuscular and Neurometabolic Disease, McMaster University, Hamilton, Ontario, Canada.

David F Wensley (DF)

Division of Pediatric Respirology, Department of Pediatrics, BC Children's Hospital, Vancouver, British Columbia, Canada.
Department of Pediatrics, The University of British Columbia, Vancouver, British Columbia, Canada.

David Zielinski (D)

Division of Pediatric Respirology, Department of Pediatrics, Montreal Children's Hospital, Montreal, Quebec, Canada.
Research Institute of the McGill University Health Centre, Montreal, Quebec, Canada.

Louise Rose (L)

Department of Midwifery and Palliative Care, King's College London Florence Nightingale School of Nursing and Midwifery, London, London, UK.
Critical Care Directorate and Lane Fox Respiratory Unit, Guy's and St Thomas' NHS Foundation Trust, London, London, UK.

Nicole Sheers (N)

Department of Respiratory and Sleep Medicine, Austin Health, Heidelberg, Victoria, Australia.
Institute for Breathing and Sleep, Heidelberg, Victoria, Australia.
Faculty of Medicine Dentistry and Health Sciences, The University of Melbourne, Melbourne, Victoria, Australia.
ORCID: 0000-0003-1847-4266

David J Berlowitz (DJ)

Department of Respiratory and Sleep Medicine, Institute for Breathing and Sleep, Heidelberg, Victoria, Australia.
Department of Physiotherapy, The University of Melbourne, Melbourne, Victoria, Australia.
ORCID: 0000-0003-2543-8722

Lisa Wolfe (L)

Department of Medicine and Neurology, Northwestern University Feinberg School of Medicine, Chicago, Illinois, USA.
Department of Respiratory Care, Shirley Ryan AbilityLab, Chicago, Illinois, USA.

Doug McKim (D)

Faculty of Medicine, University of Ottawa, Ottawa, Ontario, Canada.
Ottawa Hospital Research Institute, Ottawa, Ontario, Canada.
CANVent Respiratory Rehabilitation Services, Ottawa Hospital Rehabilitation Centre, Ottawa, Ontario, Canada.

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Classifications MeSH

questionsmedicales.fr › États, signes et symptômes pathologiques › Signes et symptômes › Signes et symptômes respiratoires › Toux › Routine lung volume recruitment in boys with...
questionsmedicales.fr › Eucaryotes › Animaux › Chordés › Vertébrés › Mammifères › Eutheria › Primates › Haplorhini › Catarrhini › Hominidae › Humains › Routine lung volume recruitment in boys with...
questionsmedicales.fr › Diagnostic › Techniques et procédures diagnostiques › Techniques de diagnostic respiratoire › Tests de la fonction respiratoire › Mesure des volumes pulmonaires › Routine lung volume recruitment in boys with...
questionsmedicales.fr › Malformations et maladies congénitales, héréditaires et néonatales › Maladies génétiques congénitales › Dystrophies musculaires › Myopathie de Duchenne › Routine lung volume recruitment in boys with...
questionsmedicales.fr › Diagnostic › Techniques et procédures diagnostiques › Techniques de diagnostic respiratoire › Tests de la fonction respiratoire › Routine lung volume recruitment in boys with...
questionsmedicales.fr › Phénomènes physiologiques respiratoires et circulatoires › Phénomènes physiologiques respiratoires › Capacité pulmonaire totale › Capacité vitale › Routine lung volume recruitment in boys with...