Efficacy of a physical activity programme combining individualized aerobic exercise and coaching to improve physical fitness in neuromuscular diseases (I'M FINE): study protocol of a randomized controlled trial.

BACKGROUND: In individuals with neuromuscular diseases (NMD), symptoms of muscle weakness, fatigue and pain may limit physical activity. Inactivity leads to reduced physical fitness, which further complicates daily life functioning. Due to inconclusive evidence regarding exercise in NMD, the optimal training approach and strategies to preserve an active lifestyle remain to be determined. The physical activity programme I'M FINE, consisting of individualized aerobic exercise to improve physical fitness and coaching to preserve an active lifestyle, was therefore developed. The primary objective of this study will be to evaluate the efficacy of the I'M FINE programme in terms of improved physical fitness in individuals with slowly progressive NMD, compared to usual care. METHODS: A multicentre, assessor-blinded, two armed, randomized controlled trial will be conducted in a sample of 90 individuals with slowly progressive NMD. Participants motivated to improve their reduced physical fitness will be randomized (ratio 1:1) to the I'M FINE intervention or usual care. The I'M FINE intervention consists of a six-month physical activity programme, including individualized home-based aerobic exercise to improve physical fitness (i.e. peak oxygen uptake), and motivational interviewing coaching (e.g. goal setting, self-management) to adopt and preserve an active lifestyle. Measurements will be performed at baseline, post-intervention, and at 12- and 18-months follow-up. The primary outcome is peak oxygen uptake (VO2 peak) directly post intervention. Main secondary outcomes are physical capacity, muscle strength, self-efficacy, daily activity, quality of life and markers of metabolic syndrome. The primary analysis compares change in VO2 peak post-intervention between the intervention and usual care group, with analysis of covariance. DISCUSSION: The I'M FINE study will provide evidence regarding the efficacy of a physical activity intervention on the physical fitness and active lifestyle over the short- and long-term in individuals with slowly progressive NMD. These outcomes could potentially improve the (inter)national guidelines for efficacy of aerobic exercise programmes and provide insight in achieving a more active lifestyle in NMD. TRIAL REGISTRATION: (5/11/2018): Netherlands Trial Register NTR7609 (retrospectively registered), https://www.trialregister.nl/trial/7344. However, the Ethics Review Committee of the Amsterdam Medical Center (AMC) approved the study protocol on 7/11/2017. No adjustments were made to the approved study protocol before the first participant enrolment and registration. Registration was done after the second participant enrolment and the information in the register corresponds one on one with the approved study protocol.

Physical strain of walking in people with neuromuscular diseases is high and relates to step activity in daily life.

OBJECTIVE: To determine the physical strain of walking and assess its relationship with daily steps and intensity of daily activity in people with neuromuscular diseases. DESIGN: Cross-sectional study. SUBJECTS/PATIENTS: Sixty-one adults with neuromuscular diseases. METHODS: Physical strain of walking, defined as oxygen consumption during comfortable walking relative to peak oxygen uptake. Daily step count and daily time spent in moderate and vigorous physical activity were assessed using accelerometry and heart rate measurements, respectively. Regression analyses assessed the relationships between log daily step count and log daily time spent in moderate and vigorous physical activity, and physical strain of walking. RESULTS: The mean (standard deviation) physical strain of walking was 73 (20)% Log daily step count and physical strain were negatively associated (ß = -0.47). No association was found with log daily time spent in moderate and vigorous physical activity. CONCLUSIONS: The highly increased physical strain of comfortable walking indicates that walking is very demanding for people with neuromuscular diseases and is associated with a reduction in daily step activity. The absence of a relationship between intensity of activities and physical strain indicates that, despite a reduction in daily step activity, strenuous daily activities may still be performed.

A Mobile Health App to Support Home-Based Aerobic Exercise in Neuromuscular Diseases: Usability Study.

BACKGROUND: Home-based aerobic exercise in people with neuromuscular diseases (NMDs) has benefits compared to exercise in the hospital or a rehabilitation center because traveling is often cumbersome due to mobility limitations, and societal costs are lower. Barriers to home-based aerobic exercise include reduced possibilities for monitoring and lack of motivation. To overcome these and other barriers, we developed a mobile health app: Keep on training with ReVi (hereafter referred to as ReVi). OBJECTIVE: We aimed to determine the usability of the ReVi app. METHODS: Patients followed a 4-month, polarized, home-based aerobic exercise program on a cycle or rowing ergometer, with 2 low-intensity sessions and 1 high-intensity session per week supported by the ReVi app. The app collected training data, including heart rate and ratings of perceived exertion, provided real-time feedback on reaching target intensity zones, and enabled monitoring via an online dashboard. Physiotherapists instructed patients on how to use the ReVi app and supervised them during their training program. Patients and physiotherapists separately evaluated usability with self-developed questionnaires, including 9 questions on a 5-point Likert scale, covering the usability elements efficiency, effectiveness, and satisfaction. RESULTS: Twenty-nine ambulatory adult patients (n=19 women; mean age 50.4, SD 14.2 years) with 11 different slowly progressive NMDs participated. Both patients and physiotherapists (n=10) reported that the app, in terms of its efficiency, was easy to use and had a rapid learning curve. Sixteen patients (55%) experienced 1 or more technical issue(s) during the course of the exercise program. In the context of effectiveness, 23 patients (81%) indicated that the app motivated them to complete the program and that it helped them to exercise within the target intensity zones. Most patients (n=19, 70%) and physiotherapists (n=6, 60%) were satisfied with the use of the app. The median attendance rate was 88% (IQR 63%-98%), with 76% (IQR 69%-82%) of time spent within the target intensity zones. Four adverse events were reported, 3 of which were resolved without discontinuation of the exercise program. CONCLUSIONS: The usability of the ReVi app was high, despite the technical issues that occurred. Further development of the app to resolve these issues is warranted before broader implementation into clinical practice.

Efficacy of aerobic exercise on aerobic capacity in slowly progressive neuromuscular diseases: A systematic review and meta-analysis.

BACKGROUND: Aerobic exercise aims to improve aerobic capacity. OBJECTIVE: To summarize the evidence on the efficacy of aerobic exercise on aerobic capacity in slowly progressive neuromuscular diseases (NMDs). METHODS: We searched the electronic databases MEDLINE, EMBASE, SPORTDiscus and Web of Science Conference Proceedings Index for articles published up to June 17, 2021, selecting randomized controlled trials that included adults with slowly progressive NMDs and compared aerobic exercise to no aerobic exercise. The primary outcome was peak oxygen uptake (VO2peak) directly post-intervention. Secondary outcomes included other peak test parameters, submaximal test parameters, long-term outcomes ≥8 weeks post-intervention, adherence and adverse events. Meta-analyses were performed for the primary outcome and for secondary outcomes when reported in more than 2 studies. Risk of bias was assessed with the Cochrane Risk of Bias tool and quality of evidence according to GRADE. RESULTS: Nine studies were included (195 participants with 8 different NMDs). Eight studies were rated at high risk of bias and 1 study was rated at some concerns. Duration of exercise programs ranged from 6 to 26 weeks, with 3 weekly training sessions of 20 to 40 min, based on maximal capacity. Meta-analyses revealed short-term moderate beneficial effects of aerobic exercise on VO2peak (standardized mean difference [SMD] 0.55, 95% CI 0.23; 0.86) and peak workload (SMD 0.61, 95% CI 0.24; 0.99). Long-term effects were not assessed. Most training sessions (83-97%) were completed, but time spent in target intensity zones was not reported. Included studies lacked detailed adverse event reporting. CONCLUSIONS: There is low-quality evidence that aerobic exercise is safe and leads to moderate improvement of aerobic capacity directly post-intervention in slowly progressive NMDs, but the long-term efficacy remains unclear. Detailed information about the time spent in target intensity zones and adverse events is lacking. PROSPERO: CRD42020200083.

Relationship Between Heart Rate and Perceived Exertion in Neuromuscular Diseases: How do Laboratory-Based Exercise Testing Results Translate to Home-Based Aerobic Training Sessions?

OBJECTIVE: To examine the heart rate-perceived exertion relationship between maximal exercise testing and home-based aerobic training in neuromuscular diseases. DESIGN: Multicentre randomized controlled trial, intervention group data. PARTICIPANTS: Individuals with Charcot-Marie-Tooth disease (n = 17), post-polio syndrome (n = 7) or other neuromuscular diseases (n = 6). METHODS: Participants followed a 4-month, homebased aerobic training programme guided by heart rate. Heart rate and ratings of perceived exertion (6-20 Borg Scale) were assessed for each minute during a maximal exercise test, and at the end of each exercise interval and recovery period during training. Heart rate and corresponding ratings of perceived exertion values of individual participants during training were visualized using plots, together with the exercise testing linear regression line between heart rate and ratings of perceived exertion. RESULTS: High correlation coefficients (i.e. > 0.70) were found between heart rate and ratings of perceived exertion, in all participants during testing (n = 30), and in 57% of the participants during training. Based on the plots the following distribution was found; participants reporting lower (n = 12), similar (n = 10), or higher (n = 8) ratings of perceived exertion values for corresponding heart rates during training compared with testing. CONCLUSION: Most participants had a different perception of effort for corresponding heart rates during training in comparison with exercise testing. Healthcare professionals should be aware that this may imply under- and over-training.

Logical validation and evaluation of practical feasibility for the SCRuM (School Clinical Rugby Measure) test battery developed for young adolescent rugby players in a resource-constrained environment.

There is a growing impetus towards usage of test batteries in talent identification (TID) programmes in rugby. Consequently, there are many test batteries in existence profiling anthropometric, physiological characteristics and rugby-specific skills. There is no consensus in the literature on the constituent variables and corresponding tests required to inform TID programs. Following development of a new test battery called the SCRuM (School Clinical Rugby Measure), this study aimed at establishing face, logical validity and practical feasibility of included tests. The test battery, initially comprised of 23 items, had its face and logical validity evaluated by five (5) adolescent rugby coaches and 20 rugby experts, respectively. Logical validation was conducted in two questionnaire-based rounds with Content Validity Index (I-CVI) calculated for each variable. Subsequently, a cross-sectional study targeting 30 local rugby coaches was conducted to determine the perceived practical feasibility of each test item. The results showed excellent I-CVI (>0.78) for 17 variables (speed, weight, height and skin fold measures, repeated high-intensity exercise performance ability, prolonged high-intensity intermittent running ability, change of direction speed, anaerobic capacity, lower-and upper body muscular power and strength, muscular flexibility, reactive agility, passing for accuracy, tackling proficiency, and catching). However, three tests, namely, Reactive Agility, One Repetition Maximum Back Squat and One Repetition Maximum Bench Press had low test-feasibility indices (T-FI< 35) suggesting practicality concerns with implementation in the Zimbabwean context. Thus, these findings suggest the need for substitution or development of new practically feasible tests for upper-and lower body muscular strength and reactive agility.

Psychometric evaluation of commonly used game-specific skills tests in rugby: A systematic review.

OBJECTIVES: To (1) give an overview of commonly used game-specific skills tests in rugby and (2) evaluate available psychometric information of these tests. METHODS: The databases PubMed, MEDLINE CINAHL and Africa Wide information were systematically searched for articles published between January 1995 and March 2017. First, commonly used game-specific skills tests were identified. Second, the available psychometrics of these tests were evaluated and the methodological quality of the studies assessed using the Consensus-based Standards for the selection of health Measurement Instruments checklist. Studies included in the first step had to report detailed information on the construct and testing procedure of at least one game-specific skill, and studies included in the second step had additionally to report at least one psychometric property evaluating reliability, validity or responsiveness. RESULTS: 287 articles were identified in the first step, of which 30 articles met the inclusion criteria and 64 articles were identified in the second step of which 10 articles were included. Reactive agility, tackling and simulated rugby games were the most commonly used tests. All 10 studies reporting psychometrics reported reliability outcomes, revealing mainly strong evidence. However, all studies scored poor or fair on methodological quality. Four studies reported validity outcomes in which mainly moderate evidence was indicated, but all articles had fair methodological quality. CONCLUSION: Game-specific skills tests indicated mainly high reliability and validity evidence, but the studies lacked methodological quality. Reactive agility seems to be a promising domain, but the specific tests need further development. Future high methodological quality studies are required in order to develop valid and reliable test batteries for rugby talent identification. TRIAL REGISTRATION NUMBER: PROSPERO CRD42015029747.

A systematic review investigating measurement properties of physiological tests in rugby.

BACKGROUND: This systematic review was conducted with the first objective aimed at providing an overview of the physiological characteristics commonly evaluated in rugby and the corresponding tests used to measure each construct. Secondly, the measurement properties of all identified tests per physiological construct were evaluated with the ultimate purpose of identifying tests with strongest level of evidence per construct. METHODS: The review was conducted in two stages. In all stages, electronic databases of EBSCOhost, Medline and Scopus were searched for full-text articles. Stage 1 included studies examining physiological characteristics in rugby. Stage 2 included studies evaluating measurement properties of all tests identified in Stage 1 either in rugby or related sports such as Australian Rules football and Soccer. Two independent reviewers screened relevant articles from titles and abstracts for both stages. RESULTS: Seventy studies met the inclusion criteria for Stage 1. The studies described 63 tests assessing speed (8), agility/change of direction speed (7), upper-body muscular endurance (8), upper-body muscular power (6), upper-body muscular strength (5), anaerobic endurance (4), maximal aerobic power (4), lower-body muscular power (3), prolonged high-intensity intermittent running ability/endurance (5), lower-body muscular strength (5), repeated high-intensity exercise performance (3), repeated-sprint ability (2), repeated-effort ability (1), maximal aerobic speed (1) and abdominal endurance (1). Stage 2 identified 20 studies describing measurement properties of 21 different tests. Only moderate evidence was found for the reliability of the 30-15 Intermittent Fitness. There was limited evidence found for the reliability and/or validity of 5 m, 10 m, 20 m speed tests, 505 test, modified 505 test, L run test, Sergeant Jump test and bench press repetitions-to-fatigue tests. There was no information from high-quality studies on the measurement properties of all the other tests identified in stage 1. CONCLUSION: A number of physiological characteristics are evaluated in rugby. Each physiological construct has multiple tests for measurement. However, there is paucity of information on measurement properties from high-quality studies for the tests. This raises questions about the usefulness and applicability of these tests in rugby and creates a need for high-quality future studies evaluating measurement properties of these physiological tests. TRIAL REGISTRATIONS: PROSPERO CRD 42015029747.