Measures of postural control and mobility during dual-tasking as candidate markers of instability in Huntington's disease

Muratori, Lisa M.; Quinn, Lori; Li, Xueyao; Youdan, Gregory; Busse, Monica; Fritz, Nora

doi:10.1016/j.humov.2021.102881

Cited by 8 publications

(5 citation statements)

References 53 publications

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“…In a cross-sectional study, compliance and psychometric properties of upper and lower limb variables (e.g., sway path, spiral drawing speed variability, median turn speed, and step frequency variance) were extracted from a digital monitoring platform [ 55 ]. Balance and body sway (as root mean square [ 61 , 64 ], jerk, sway area and power [ 65 ], peak and mean thoracic and pelvic excursion [ 66 ]) were impaired even at the premanifest stage of disease.…”

Section: Resultsmentioning

confidence: 99%

The use of digital outcome measures in clinical trials in rare neurological diseases: a systematic literature review

Poleur

Markati

Servais

2023

Orphanet J Rare Dis

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Developing drugs for rare diseases is challenging, and the precision and objectivity of outcome measures is critical to this process. In recent years, a number of technologies have increasingly been used for remote monitoring of patient health. We report a systematic literature review that aims to summarize the current state of progress with regard to the use of digital outcome measures for real-life motor function assessment of patients with rare neurological diseases. Our search of published literature identified 3826 records, of which 139 were included across 27 different diseases. This review shows that use of digital outcome measures for motor function outside a clinical setting is feasible and employed in a broad range of diseases, although we found few outcome measures that have been robustly validated and adopted as endpoints in clinical trials. Future research should focus on validation of devices, variables, and algorithms to allow for regulatory qualification and widespread adoption.

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Section: Resultsmentioning

confidence: 99%

The use of digital outcome measures in clinical trials in rare neurological diseases: a systematic literature review

Poleur

Markati

Servais

2023

Orphanet J Rare Dis

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“…However, despite utilizing differing classification systems for disease severity, two included studies [ 2 , 14 ] found preliminary evidence that spatiotemporal gait changes worsen with each stage of the disease. HHT CAG repeat length can also be used to classify disease severity [ 42 - 47 ]. With less than half of the included studies reporting these data, the association of CAG repeat length with gait parameter changes remains unclear.…”

Section: Discussionmentioning

confidence: 99%

“…HHT CAG repeat length can also be used to classify disease severity. [42][43][44][45][46][47] With less than half of the included studies reporting these data, the association of CAG repeat length with gait parameter changes remains unclear. If individual patient data from the original studies are available, it may be possible to perform further data synthesis and meta-analyses on gait parameters with predefined subgroups that would allow the exploration of the relationship among gait parameters, CAG repeat length and predominant movement disorder.…”

Section: Discussionmentioning

confidence: 99%

“…Andrzejewski et al 43 Anwar et al 26 Beckmann et al 1 Bilney et al 29 Churchyard et al 32 Collett et al 2 Dalton et al 21 Danoudis and Iansek 44 de Tommaso et al 45 Delval et al 28 Delval et al 30 Delval et al 22 Desai et al 46 Gaßner et al 9 Grimbergen et al 3 Hausdorff et al 11 Hausdorff et al 10 Koller and Trimble 24 Muratori et al 47 Purcell et al 31 Pyo et al 25 Radovanović et al 27 Rao et al 8 Rao et al 14 Reynolds et al 23 Figure 1. Quality analysis of the included studies using the Joanna Briggs Institute's checklist for analytical cross-sectional studies and supplementary assessments.…”

Section: Quantitative Data Synthesismentioning

confidence: 99%

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Spatiotemporal Gait Parameters in Adults With Premanifest and Manifest Huntington’s Disease: A Systematic Review

Browning,

Holland,

Wellwood

et al. 2023

JMD

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ObjectiveaaTo systematically review and critically evaluate literature on spatiotemporal gait deviations in individuals with premanifest and manifest Huntington’s Disease (HD) in comparison with healthy cohorts.MethodsaaWe conducted a systematic review, guided by the Joanna Briggs Institute’s Manual for Evidence Synthesis and pre-registered with the International Prospective Register of Systematic Reviews. Eight electronic databases were searched. Studies comparing spatiotemporal footstep parameters in adults with premanifest and manifest HD to healthy controls were screened, included and critically appraised by independent reviewers. Data on spatiotemporal gait changes and variability were extracted and synthesised. Meta-analysis was performed on gait speed, cadence, stride length and stride length variability measures.ResultsaaWe screened 2,721 studies, identified 1,245 studies and included 25 studies (total 1,088 participants). Sample sizes ranged from 14 to 96. Overall, the quality of the studies was assessed as good, but reporting of confounding factors was often unclear. Meta-analysis found spatiotemporal gait deviations in participants with HD compared to healthy controls, commencing in the premanifest stage. Individuals with premanifest HD walk significantly slower (-0.17 m/s; 95% confidence interval [CI] [-0.22, -0.13]), with reduced cadence (-6.63 steps/min; 95% CI [-10.62, -2.65]) and stride length (-0.09 m; 95% CI [-0.13, -0.05]). Stride length variability was also increased in premanifest cohorts by 2.18% (95% CI [0.69, 3.68]), with these changes exacerbated in participants with manifest disease.ConclusionaaFindings suggest individuals with premanifest and manifest HD display significant spatiotemporal footstep deviations. Clinicians could monitor individuals in the premanifest stage of disease for gait changes to identify the onset of Huntington’s symptoms.

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“…Thus, therapists may wish to include an assessment of dual-task [ 17, 20–26 ] in addition to the core set of recommended outcomes ( Fig. 1 ) to monitor dual-task performance over time; our recent work suggests that the TUG Cognitive may be a sensitive measure of dual-task walking in HD [ 20 ]. Providing clear instructions and written materials and including the caregiver and/or family in education can help to overcome this barrier throughout the disease course.…”

Section: Special Considerations In the Physical Therapy Management Of...mentioning

confidence: 99%

Clinical Decision Trees to Guide Physical Therapy Management of Persons with Huntington’s Disease

Fritz

Kegelmeyer

Rao

et al. 2022

JHD

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Background: In 2020, our group published physical therapy clinical practice guidelines (CPG) for people with Huntington’s disease (HD). The guideline recommendations were categorized according to six primary movement impairment classifications. Objective: To facilitate implementation of this CPG, we have developed guideline-based algorithms for physical therapy assessments and interventions and recommendations for therapists to overcome barriers to CPG implementation for people with HD. Methods: We conducted a literature review of papers that evaluated physical therapy interventions in individuals with HD (n = 26) to identify assessments for each of the primary movement impairment classifications, and then searched for papers (n = 28) that reported their clinometric/psychometric properties in HD. Assessments were evaluated using modified Movement Disorder Society Committee on Rating Scales criteria and other relevant criteria. Results: We identified a “core set” of physical therapy assessments for persons with HD, including the Six Minute Walk Test, Timed Up and Go Test, Berg Balance Scale, and the Medical Outcomes Study Short Form 36 (SF-36). We then developed guideline-based decision trees to assist in decision making and implementation of the CPG into practice for persons with HD across the continuum of care. Finally, we developed strategies for overcoming barriers to implementation, such as seeking specialized training in HD, engaging caretakers or family members to help the person with HD to exercise, and establishing clinical pathways that support early physical therapy referrals. Conclusion: Knowledge translation documents such as this are essential to promoting implementation of the physical therapy CPGs into clinical practice.

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Measures of postural control and mobility during dual-tasking as candidate markers of instability in Huntington's disease

Cited by 8 publications

References 53 publications

The use of digital outcome measures in clinical trials in rare neurological diseases: a systematic literature review

The use of digital outcome measures in clinical trials in rare neurological diseases: a systematic literature review

Spatiotemporal Gait Parameters in Adults With Premanifest and Manifest Huntington’s Disease: A Systematic Review

Clinical Decision Trees to Guide Physical Therapy Management of Persons with Huntington’s Disease

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