Publication related to RSI or an RSI staff member
Routine lung volume recruitment in boys with Duchenne muscular dystrophy: a randomised clinical trial.
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.
Authors
- Katz, 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.
- Mah, 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.
- McMillan, 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.
- Campbell, 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.
- Bijelic, Vid, Bijelic V, CHEO Research Institute, Ottawa, Ontario, Canada.
- Barrowman, Nick, Barrowman N, CHEO Research Institute, Ottawa, Ontario, Canada.; Faculty of Medicine, University of Ottawa, Ottawa, Ontario, Canada.
- Momoli, Franco, Momoli F, Faculty of Medicine, University of Ottawa, Ottawa, Ontario, Canada.
- Blinder, Henrietta, Blinder H, CHEO Research Institute, Ottawa, Ontario, Canada.
- Aaron, 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.
- McAdam, Laura C, McAdam LC, Holland Bloorview Kids Rehabilitation Hospital, Toronto, Ontario, Canada.; Department of Pediatrics, University of Toronto, Toronto, Ontario, Canada.
- Nguyen, The Thanh Diem, Nguyen TTD, Department of Respiratory Medicine, Centre Hospitalier Universitaire Sainte-Justine, Montreal, Quebec, Canada.
- Tarnopolsky, Mark, Tarnopolsky M, Division of Neuromuscular and Neurometabolic Disease, McMaster University, Hamilton, Ontario, Canada.
- Wensley, 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.
- Zielinski, 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.
- Rose, 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.
- Sheers, 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.
- Berlowitz, 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.
- Wolfe, 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.
- McKim, 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.
YEAR OF PUBLICATION: 2022
SOURCE: Thorax. 2022 Aug;77(8):805-811. doi: 10.1136/thoraxjnl-2021-218196. Epub 2022 Mar 2.
JOURNAL TITLE ABBREVIATION: Thorax
JOURNAL TITLE: Thorax
ISSN: 1468-3296 (Electronic) 0040-6376 (Print) 0040-6376 (Linking)
VOLUME: 77
ISSUE: 8
PAGES: 805-811
PLACE OF PUBLICATION: England
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.
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.
COPYRIGHT INFORMATION: (c) 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.
LANGUAGE: eng
DATE OF PUBLICATION: 2022 Aug
DATE OF ELECTRONIC PUBLICATION: 20220302
DATE COMPLETED: 20220727
DATE REVISED: 20240901
MESH DATE: 2022/07/28 06:00
EDAT: 2022/03/04 06:00
STATUS: MEDLINE
PUBLICATION STATUS: ppublish
LOCATION IDENTIFIER: 10.1136/thoraxjnl-2021-218196 [doi]
SECONDARY SOURCE ID: ClinicalTrials.gov/NCT01999075
COMMENT IN:
OWNER: NLM
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