Short summary

FRAME-EX explores how FRAME Running-a sport using a three-wheeled running frame-affects physical function, quality of life, and participation in everyday life among children and youth with disabilities. Using mixed methods, the project includes a feasibility study, an intervention study, and qualitative interviews, aiming to generate practical knowledge for inclusive physical activity.

Rationale

BACKGROUND Children and young people with physical disabilities, including cerebral palsy (CP) and muscular dystrophy, are less physical active and socially engaged compared to those without disabilities and have low levels of daily physical activity, which may lead to decreased health status, higher risk of developing secondary impairments in the musculoskeletal and/or cardio-respiratory systems. Such impairments may contribute to a vicious circle leading to further deconditioning and lower life expectancy, with respiratory illness being the leading cause of death. Current evidence points toward the fact that many of these changes may be preventable or reversible by a physical active lifestyle and that participating in physical activity is beneficial for quality of life among children and adolescents with a disability. As children and young people with moderate or severe impairments cannot engage in independent running and cycling, alternative, enjoyable, and playful training modes are needed. Frame Running, an emerging yet scientifically overlooked activity developed in Denmark in 1991, is an adaptive sport for individuals with mobility impairments. It involves using a three-wheeled frame, equipped with a saddle and handlebars but no pedals, allowing the athlete to run or walk using their feet while being supported by the frame. This sport has become popular among children and young people with CP as it offers recreational and competitive opportunities for those with moderate to severe walking limitations. An ongoing scoping review within our research group is currently mapping the existing quantitative and qualitative research on Frame Running. Preliminary results shows that only 15 studies have been published in this area, with 9 focusing on the health impacts of Frame Running. A previous intervention study, without a control group, demonstrated that a 12-week research-initiated Frame Running program twice a week led to improvements in cardiorespiratory and peripheral muscle adaptations in children and young adults with CP. Moreover, a cross-sectional study and an interview study in children and young people with CP and other disabilities found a positive impact on psychosocial factors and self-reported quality of life, respectively. Noteworthy, no studies have evaluated potential positive outcomes of Frame Running activities in a pragmatic real-world setting, and the generalizability of current research in a societal sense is non-existing. Regarding ongoing and/or future studies there is only one feasibility study and one multi-center randomized controlled trial (RCT) registered. Both studies primarily focus on cardiovascular fitness, employing a standardized research-initiated intervention program partially tailored to individual needs. There is, therefore, a need to investigate the impact and implications of Frame Running participation in children and young people within a pragmatic and real-world context, with functional ability and quality of life as primary focus areas. AIMS Within the framework of a combined-method evaluation design, the overall purpose of the PhD project is to investigate how the implementation of a pragmatic Frame Running intervention affect functional ability, quality of life, and participation in children and young people with physical disabilities. Study 1; to study the feasibility of testing (evaluability assessment) to be used in a following intervention study in a pragmatic setting for improving functional ability and quality of life in children and young people with disabilities. Study 2a; to evaluate whether a 12-weeks Frame Running intervention (added to everyday care) improves specific domains of functional ability and quality of life compared with a prior control-period without Frame Running activities, and study 2b; to explore how Frame Running activities impact the sense of participation and everyday living in children and young people with disabilities, as well as their families.

Description of the cohort

Children and young people with physical disabilities, age 8-18 years, from 4 Frame Running clubs in Odense, Randers, Kolding and Frederiksberg.

Data and biological material

Participant characteristic (age, sex, diagnosis, weight, height, assistive devices) Questionnaire data (PEDI-CAT, pedsql, CAPE-PAC, PIADS), physical tests (6 minutes frame running test), 1 minute walk-test, 1 min sit-to-stand test. Semistructured interview

Collaborating researchers and departments

Health Sciences Research Centre & Department of Physiotherapy.

• Camilla Marie Larsen

Research and Implementation Centre for Human Movement and Learning, University of Southern Denmark

• Lars Breum Christiansen
• Andreas Jørgensen

Department of Regional Health Research, University of Southern Denmark

• Helle Mätzke Rasmussen

Department of Clinical Research, University of Southern Denmark

• Anders Holsgaard Larsen