Interventions for promoting physical activity in people with neuromuscular disease

Katherine Jones, Fiona Hawke, Jane Newman, James Al Miller, Joshua Burns, Djordje G Jakovljevic, Grainne Gorman, Douglass M Turnbull, Gita Ramdharry

    Research output: Contribution to journalReview articlepeer-review

    12 Citations (Scopus)

    Abstract

    BACKGROUND: The World Health Organization (WHO) recommends that people of all ages take regular and adequate physical activity. If unable to meet the recommendations due to health conditions, international guidance advises being as physically active as possible. Evidence from community interventions of physical activity indicate that people living with medical conditions are sometimes excluded from participation in studies. In this review, we considered the effects of activity-promoting interventions on physical activity and well-being in studies, as well as any adverse events experienced by participants living with inherited or acquired neuromuscular diseases (NMDs). OBJECTIVES: To assess the effects of interventions designed to promote physical activity in people with NMD compared with no intervention or alternative interventions.

    SEARCH METHODS: On 30 April 2020, we searched Cochrane Neuromuscular Specialised Register, CENTRAL, Embase, MEDLINE, and ClinicalTrials.Gov. WHO ICTRP was not accessible at the time.

    SELECTION CRITERIA: We considered randomised or quasi-randomised trials, including cross-over trials, of interventions designed to promote physical activity in people with NMD compared to no intervention or alternative interventions. We specifically included studies that reported physical activity as an outcome measure. Our main focus was studies in which promoting physical activity was a stated aim but we also included studies in which physical activity was assessed as a secondary or exploratory outcome.

    DATA COLLECTION AND ANALYSIS: We used standard Cochrane procedures.

    MAIN RESULTS: The review included 13 studies (795 randomised participants from 12 studies; number of participants unclear in one study) of different interventions to promote physical activity. Most studies randomised a minority of invited participants. No study involved children or adolescents and nine studies reported minimal entry criteria for walking. Participants had one of nine inherited or acquired NMDs. Types of intervention included structured physical activity support, exercise support (as a specific form of physical activity), and behaviour change support that included physical activity or exercise. Only one included study clearly reported that the aim of intervention was to increase physical activity. Other studies reported or planned to analyse the effects of intervention on physical activity as a secondary or exploratory outcome measure. Six studies did not report results for physical activity outcomes, or the data were not usable. We judged 10 of the 13 included studies at high or unclear risk of bias from incomplete physical activity outcome reporting. We did not perform a meta-analysis for any comparison because of differences in interventions and in usual care. We also found considerable variation in how studies reported physical activity as an outcome measure. The studies that reported physical activity measurement did not always clearly report intention-to-treat (ITT) analysis or whether final assessments occurred during or after intervention. Based on prespecified measures, we included three comparisons in our summary of findings. A physical activity programme (weight-bearing) compared to no physical activity programme One study involved adults with diabetic peripheral neuropathy (DPN) and reported weekly duration of walking during and at the end of a one-year intervention using a StepWatch ankle accelerometer. Based on the point estimate and low-certainty evidence, intervention may have led to an important increase in physical activity per week; however, the 95% confidence interval (CI) included the possibility of no difference or an effect in either direction at three months (mean difference (MD) 34 minutes per week, 95% CI -92.19 to 160.19; 69 participants), six months (MD 68 minutes per week, 95% CI -55.35 to 191.35; 74 participants), and 12 months (MD 49 minutes per week, 95% CI -75.73 to 173.73; 70 participants). Study-reported effect estimates for foot lesions and full-thickness ulcers also included the possibility of no difference, a higher, or lower risk with intervention. A sensor-based, interactive exercise programme compared to no sensor-based, interactive exercise programme One study involved adults with DPN and reported duration of walking over 48 hours at the end of four weeks' intervention using a t-shirt embedded PAMSys sensor. It was not possible to draw conclusions about the effectiveness of the intervention from the very low-certainty evidence (MD -0.64 hours per 48 hours, 95% CI -2.42 to 1.13; 25 participants). We were also unable to draw conclusions about impact on the Physical Component Score (PCS) for quality of life (MD 0.24 points, 95% CI -5.98 to 6.46; 35 participants; very low-certainty evidence), although intervention may have made little or no difference to the Mental Component Score (MCS) for quality of life (MD 5.10 points, 95% CI -0.58 to 10.78; 35 participants; low-certainty evidence). A functional exercise programme compared to a stretching exercise programme One study involved adults with spinal and bulbar muscular atrophy and reported a daily physical activity count at the end of 12 weeks' intervention using an Actical accelerometer. It was not possible to draw conclusions about the effectiveness of either intervention (requiring compliance) due to low-certainty evidence and unconfirmed measurement units (MD -8701, 95% CI -38,293.30 to 20,891.30; 43 participants). Functional exercise may have made little or no difference to quality of life compared to stretching (PCS: MD -1.10 points, 95% CI -5.22 to 3.02; MCS: MD -1.10 points, 95% CI -6.79 to 4.59; 49 participants; low-certainty evidence). Although studies reported adverse events incompletely, we found no evidence of supported activity increasing the risk of serious adverse events.

    AUTHORS' CONCLUSIONS: We found a lack of evidence relating to children, adolescents, and non-ambulant people of any age. Many people living with NMD did not meet randomised controlled trial eligibility criteria. There was variation in the components of supported activity intervention and usual care, such as physical therapy provision. We identified variation among studies in how physical activity was monitored, analysed, and reported. We remain uncertain of the effectiveness of promotional intervention for physical activity and its impact on quality of life and adverse events. More information is needed on the ITT population, as well as more complete reporting of outcomes. While there may be no single objective measure of physical activity, the study of qualitative and dichotomous change in self-reported overall physical activity might offer a pragmatic approach to capturing important change at an individual and population level.

    Original languageEnglish
    Article numberCD013544
    Number of pages126
    JournalThe Cochrane Library
    Volume2021
    Issue number5
    Early online date24 May 2021
    DOIs
    Publication statusPublished - 24 May 2021

    Funder

    Funding Information: Aase and Einar Danielsens Foundation, Augustinus Foundation, and AP Moeller Foundation provided financial support. Funding Information: JN: none. She was involved in Okkersen 2018. This study was funded by the European Union Seventh Framework Program, under grant agreement number 305697 (the Observational Prolonged Trial In Myotonic dystrophy type 1 to Improve quality of life Standards, a Target Identification Collaboration (OPTIMISTIC) project).

    Funding Information: The National Institutes of Health (grant nos. NCMRR R21 HD058938, T32 HD007434-17 NSMRC R24HD650837, NIH UL1 RR024992), Diabetes Research Training Center (grant number 5 P60 DK20579), and scholarships from the Foundation for Physical Therapy.

    Funding Information: Intramural research funds from the National Institute of Neurological Disorders and Stroke.

    Funding Information: GBS/CIDP International also known as GAIN charity (Guillain-Barré and Associated Inflammatory Neuropathies, UK support group, ref: GBS2011/8).

    Additional funding support was received from London South CLRN contingency funds.

    Funding Information: Prinses Beatrix SpierFonds (The Dutch Public Fund for Neuromuscular Disorders), the Netherlands Organisation for Health Research and Development (ID: ZonMW 89000003), and the FSHD Global Research Foundation.

    Funding Information: JB: receives research funding from NIH (National Institute of Neurological Diseases and Stroke and National Center for Advancing Translational Sciences, Inherited Neuropathies Consortium, Rare Disease Clinical Research Network #2U54NS065712), Charcot-Marie Tooth Association of Australia, Charcot-Marie Tooth Association (USA), Diabetes Australia, Multiple Sclerosis Research Australia, Sydney Southeast Asia Centre, New Zealand Neuromuscular Research Foundation Trust, Elizabeth Lottie May Rosenthal Bone Bequest and Perpetual Limited.

    Funding Information: DMT: our work was supported by The Wellcome Trust Centre for Mitochondrial Research (906919), the Newcastle University Centre for Brain Ageing and Vitality supported by Biotechnology and Biological Sciences Research Council, Engineering and Physical Sciences Research Council, Economic and Social Research Council, and Medical Research Council as part of the cross-council Lifelong Health and Wellbeing Initiative (G0700718).

    ASJC Scopus subject areas

    • Pharmacology (medical)

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