Abstract
Background Task-specific training may be a suitable intervention to address mobility limitations in people with Huntington disease (HD).
Objective The aim of this study was to assess the feasibility and safety of goal-directed, task-specific mobility training for individuals with mid-stage HD.
Design This study was a randomized, blinded, feasibility trial; participants were randomly assigned to control (usual care) and intervention groups.
Setting This multisite study was conducted in 6 sites in the United Kingdom.
Patients Thirty individuals with mid-stage HD (13 men, 17 women; mean age=57.0 years, SD=10.1) were enrolled and randomly assigned to study groups.
Intervention Task-specific training was conducted by physical therapists in participants' homes, focusing on walking, sit-to-stand transfers, and standing, twice a week for 8 weeks. Goal attainment scaling was used to individualize the intervention and monitor achievement of personal goals.
Measurements Adherence and adverse events were recorded. Adjusted between-group comparisons on standardized outcome measures were conducted at 8 and 16 weeks to determine effect sizes.
Results Loss to follow-up was minimal (n=2); adherence in the intervention group was excellent (96.9%). Ninety-two percent of goals were achieved at the end of the intervention; 46% of the participants achieved much better than expected outcomes. Effect sizes on all measures were small.
Limitations Measurements of walking endurance were lacking.
Conclusions The safety of and excellent adherence to a home-based, task-specific training program, in which most participants exceeded goal expectations, are encouraging given the range of motivational, behavioral, and mobility issues in people with HD. The design of the intervention in terms of frequency (dose), intensity (aerobic versus anaerobic), and specificity (focused training on individual tasks) may not have been sufficient to elicit any systematic effects. Thus, a larger-scale trial of this specific intervention does not seem warranted.
Footnotes
Dr Quinn, Professor Dawes, Professor Rosser, and Dr Busse provided concept/idea/research design. Dr Quinn, Professor Dawes, Dr Kelson, Ms Townson, and Dr Busse provided writing. Ms Debono, Dr Rickards, Professor Tabrizi, Dr Quarrell, Dr Trender-Gerhard, and Dr Busse provided data collection. Dr Quinn, Dr Kelson, and Ms Townson provided data analysis. Dr Quinn, Ms Debono, Ms Townson, and Dr Busse provided project management. Dr Quinn, Professor Dawes, and Dr Busse provided fund procurement. Professor Dawes, Professor Rosser, Dr Nemeth, Dr Rickards, Professor Tabrizi, and Dr Quarrell provided participants. Professor Dawes, Dr Nemeth, Dr Rickards, Professor Tabrizi, and Dr Quarrell provided facilities/equipment. Dr Rickards and Dr Quarrell provided institutional liaisons. Professor Dawes, Professor Rosser, Dr Nemeth, Dr Rickards, Professor Tabrizi, Dr Quarrell, Dr Trender-Gerhard, Ms Debono, and Ms Townson provided consultation (including review of the manuscript before submission).
The authors acknowledge all of the participants in the trial and their family members and caregivers. The authors thank all of the site staff who tirelessly worked on this trial. The authors acknowledge the European Huntington's Disease Network (EHDN), NISCHR Clinical Research Centre (NISCHR CRC) in Wales, and Dementias and Neurodegenerative Diseases Research Network (DeNDRoN) for their adoption and support of this trial. The authors also thank the members of the TRAIN-HD Steering Committee for their invaluable guidance and support: Professor Stephen Dunnett (Chair), Mrs Jacqueline Peacock, Mrs Adele Griffiths, Dr Valentina Tomasini, and Mrs Angela Hall.
Research funding for this trial was from the Huntington's Disease Association of England and Wales. Professor Dawes is funded by the Elizabeth Casson Trust and the NIHR Oxford BRC.
The project was approved by the Southeast Wales NHS Research Committee.
Members of the Task-Related Training in Huntington's Disease (TRAIN-HD) project group: Birmingham: Shabana Akhtar, Jennifer Crooks, Jennifer De Souza, Kerry Gibson, Claire Jones; Cardiff: Catherine Johnston, Karen Jones, Duncan McLauchlan; London: Joanna Allen, Rosie Cosh, Salman Haider, Monica Lewis, Nicola Robertson, Anne Rodger, Rhiannon Stokes, Rachel Taylor; Manchester: Judith Bek, Dawn Rogers, Cheryl Stopford, Jo Teal; Oxford: Johnny Collett, Loretta Davies, Bryony Sheridan; Sheffield: Laura Barnes, Kay Crossland, Rachel Cruise, Stuart Ingram, Natalie Jones, Molly Sandhu, Cat Taylor.
The trial was registered with the International Standard Randomized Controlled Trial (clinical registration number: ISRCTN94284668).
- Received March 23, 2014.
- Accepted June 28, 2014.
- © 2014 American Physical Therapy Association
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