Thesis

114 Chapter 7 can be given why results would turn out differently. More importantly, the challenge will be to investigate which (supervised) guidance and intervention protocol will be able to keep people with HSP physically active across their life span and, thus, have actual impact on their disease progression. Chronic fatigue is often a barrier for many neurological patients to become or stay physically active. This is why a multidisciplinary intervention has been developed at Radboud University Medical Center for people with neurological conditions and chronic fatigue, called “Energetic”. This 16-week multidisciplinary day-care (1-2 times per week) group program comprises individually tailored gradually increasing physical exercises, education about exercise training, adopting energy conservation techniques, and creating real-life changes in (sports) activities to prevent relapse after cessation of the program. Energetic adopts various selfmanagement principles and has proven its effectiveness on societal participation (assessed by individual goal setting with the Canadian Occupational Performance Measure) in people with various neuromuscular disorders. Energetic also proved to be cost-effective compared to usual care. 48 It is likely that people with HSP who are chronically fatigued respond equally well to a program like Energetic, but the specific frequency and content of this program would need to be adapted. This would be an interesting direction for future research. Methodological considerations In this thesis, we evaluated the efficacy of a novel gait adaptability training in people with HSP through an RCT. Some limitations related to the specific intervention and outcome measures have already been mentioned in Chapter 5, including the relatively short training period of five weeks, the absence of booster sessions, a potential ceiling effect on our primary walking adaptability outcome, and the lack of outcome measures that reflect gait performance and gait perception. Here, I consider some limitations from a broader perspective. In line with other studies, we used a five-week intervention period to evaluate the efficacy of a novel type of training. It is, however, questionable whether such a short, predetermined training period can make a difference in patient populations with progressive conditions who slowly build up their gait problems across many years. To be able to make a true impact, group differences in training activity should probably be maintained for many years as well, for instance as short physiotherapy-guided ‘booster sessions’ complemented with self-exercises at home in between. RCTs are generally accepted to generate data to acquire the highest level of scientific evidence for clinical interventions. They are conducted prospectively while their risk of bias is reduced by random group allocation. 49 On the downside, RCTs are often costly and time-consuming, and they require a sufficiently large sample size in a relatively short period of time to reach the necessary power for statistical analysis.

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