Jane Goodwin1, Jan Lecouturier1, Anna Basu2,3, Allan Colver1, Sarah Crombie4, Johanna Smith1, Denise Howel1, Elaine McColl1, Jeremy R Parr2,5, Niina Kolehmainen1,3, Andrew Roberts6, Keith Miller6, Jill Cadwgan2,7. 1. Institute of Health & Society, Newcastle University, Newcastle upon Tyne, UK. 2. Institute of Neuroscience, Newcastle University, Newcastle upon Tyne, UK. 3. Newcastle upon Tyne Hospitals NHS Foundation Trust, Newcastle upon Tyne, UK. 4. Sussex Community NHS Foundation Trust, Chailey Clinical Services, Sussex, UK. 5. Great North Children's Hospital, Newcastle upon Tyne Hospitals NHS Foundation Trust, Newcastle upon Tyne, UK. 6. Robert Jones and Agnes Hunt Orthopaedic and District Hospital NHS Trust, Oswestry, UK. 7. Evelina London Children's Hospital, Guys and St Thomas' NHS Foundation Trust, King's Health Partners, London, UK.
Abstract
BACKGROUND: Standing frames are recommended as part of postural management for young people with cerebral palsy (CP) Gross Motor Function Classification System (GMFCS) level IV or V. They may have a variety of benefits, including improving bone mineral density, gastrointestinal function and social participation. The NHS needs to know if these benefits are real, given the cost implications of use and the reported negative effects (e.g. pain). The lack of evidence for the clinical effectiveness of standing frames demonstrates the need for evaluative research. OBJECTIVE(S): The aim of the study was to explore the acceptability of a future trial to determine the clinical effectiveness of standing frames. DESIGN: A sequential mixed-methods design was used. The findings of each stage informed the next stage. We conducted surveys, focus groups and in-depth interviews. PARTICIPANTS: Professionals who work with young people who use standing frames and parents who have a child who uses a standing frame took part in a survey of current standing frame practice (n = 551), a series of focus groups (seven focus groups, 49 participants in total) and a survey of research trial acceptability and feasibility (n = 585). Twelve young people who use a standing frame were interviewed. RESULTS: Standing frames were widely used as part of postural management for young people with CP both in school and at home but more frequently in school, and particularly by young people in primary school. Achieving the prescribed use was not always possible owing to resources, environment and family factors. Participation and activity engagement were important to young people. The majority of participants believed that standing frames research is necessary. Some reported concern that stopping standing frame use for a trial would cause irreversible damage. The maximum amount of time most health professionals and parents would agree to suspend standing frame use would be 12 weeks. LIMITATIONS: Owing to the nature of recruitment, we could not calculate response rates or determine non-response bias. Therefore, participants may not be representative of all standing frame users. CONCLUSIONS: Although parents and professionals who engaged in the qualitative aspect of this research and stakeholders who took part in the design workshops appreciated the lack of clinical evidence, our surveys, qualitative information and PPI demonstrated that most people had strong beliefs regarding the clinical effectiveness of standing frames. However, with key stakeholder engagement and careful planning, a trial would be acceptable. FUTURE WORK: We recommend a carefully planned trial that includes a pilot phase. The trial should evaluate the following question: 'does using a standing frame in school improve patient-reported outcomes of participation (primary outcome), quality of life, subjective well-being, body function and body structure (secondary outcomes) in young children (aged 4-11 years) with CP GMFCS III-V?'. FUNDING: The National Institute for Health Research Health Technology Assessment programme.
RCT Entities:
BACKGROUND: Standing frames are recommended as part of postural management for young people with cerebral palsy (CP) Gross Motor Function Classification System (GMFCS) level IV or V. They may have a variety of benefits, including improving bone mineral density, gastrointestinal function and social participation. The NHS needs to know if these benefits are real, given the cost implications of use and the reported negative effects (e.g. pain). The lack of evidence for the clinical effectiveness of standing frames demonstrates the need for evaluative research. OBJECTIVE(S): The aim of the study was to explore the acceptability of a future trial to determine the clinical effectiveness of standing frames. DESIGN: A sequential mixed-methods design was used. The findings of each stage informed the next stage. We conducted surveys, focus groups and in-depth interviews. PARTICIPANTS: Professionals who work with young people who use standing frames and parents who have a child who uses a standing frame took part in a survey of current standing frame practice (n = 551), a series of focus groups (seven focus groups, 49 participants in total) and a survey of research trial acceptability and feasibility (n = 585). Twelve young people who use a standing frame were interviewed. RESULTS: Standing frames were widely used as part of postural management for young people with CP both in school and at home but more frequently in school, and particularly by young people in primary school. Achieving the prescribed use was not always possible owing to resources, environment and family factors. Participation and activity engagement were important to young people. The majority of participants believed that standing frames research is necessary. Some reported concern that stopping standing frame use for a trial would cause irreversible damage. The maximum amount of time most health professionals and parents would agree to suspend standing frame use would be 12 weeks. LIMITATIONS: Owing to the nature of recruitment, we could not calculate response rates or determine non-response bias. Therefore, participants may not be representative of all standing frame users. CONCLUSIONS: Although parents and professionals who engaged in the qualitative aspect of this research and stakeholders who took part in the design workshops appreciated the lack of clinical evidence, our surveys, qualitative information and PPI demonstrated that most people had strong beliefs regarding the clinical effectiveness of standing frames. However, with key stakeholder engagement and careful planning, a trial would be acceptable. FUTURE WORK: We recommend a carefully planned trial that includes a pilot phase. The trial should evaluate the following question: 'does using a standing frame in school improve patient-reported outcomes of participation (primary outcome), quality of life, subjective well-being, body function and body structure (secondary outcomes) in young children (aged 4-11 years) with CP GMFCS III-V?'. FUNDING: The National Institute for Health Research Health Technology Assessment programme.
Authors: Javier Güeita-Rodríguez; Anna Ogonowska-Slodownik; Natalia Morgulec-Adamowicz; Mar Lledó Martín-Prades; Juan Nicolás Cuenca-Zaldívar; Domingo Palacios-Ceña Journal: Int J Environ Res Public Health Date: 2021-03-18 Impact factor: 3.390