Introduction: To assess the effects of an intervention, the use of outcome measures relevant to the population participating in the intervention is crucial. However, these measures are primarily selected by physiotherapists and researchers, often failing to capture the main desires and expectations of children and their families. Developing a Core Outcome Set (COS) can enhance the efficiency, relevance, and overall quality of clinical research conducted in children with Cerebral Palsy (CP).
Aim: Therefore, the aim of this study is to develop a COS for telehealth physiotherapy interventions in children with CP that includes the child's and family's interests.
Methods: This study will consist of developing a COS to evaluate outcomes of telehealth physiotherapy interventions in children with CP. The following phases will be conducted: (1) a scoping review to generate a categorized list of potential outcomes for inclusion in the COS; (2) a consensus-building process involving key stakeholders involved in the care and/or research of children with CP (i.e., physiotherapists, researchers, and parents of children with CP) to prioritize the outcomes for inclusion in the COS; and (3) a consensus meeting to refine and finalize the COS. A validation phase with a separate group of parents of children with CP will also be conducted. Researchers, physiotherapists, and parents of children with CP will rate the importance of candidate outcomes on a 5-point Likert scale. Outcomes rated as "important" or "very important" by the majority (=85%) of respondents will be voted on during a consensus meeting.
Conclusion: The development and implementation of an evidence-based, consensus-driven COS for use in telehealth physiotherapy intervention studies in children with CP will help reduce heterogeneity across studies and facilitate evidence-based decision-making.
Isabella Saraiva Christovão1*, Paula Silva de Carvalho Chagas2, Hércules Ribeiro Leite1, Ana Cristina Resende Camargos1
1 Graduate Program in Rehabilitation Sciences, Universidade Federal de Minas Gerais, Belo Horizonte, Brazil
2 Graduate Program in Rehabilitation Sciences and Physical and Functional Performance, Universidade Federal de Juiz de Fora, Juiz de Fora, Brazil
*Principal investigator
Disease Category: Child health
Disease Name: Cerebral palsy
Age Range: 0 - 12
Sex:
Nature of Intervention: Physiotherapy
- Clinical experts
- Consumers (caregivers)
- Families
- Researchers
- COS for clinical trials or clinical research
- COS for practice
- Consensus meeting
- Survey
The results of the review showed that studies more frequently reported outcomes related to the child's capacity (76.6%), only 12.8% of the studies reported outcomes related to performance, which refer to the outcomes achieved by the child in their everyday life. There is a wide range of standardized instruments available to assess intervention outcomes; however, these are primarily selected by physiotherapists and researchers, often without capturing the key desires and expectations of children and their families. Current studies demonstrate that current models encompassing family-centered practice aim to maximize child outcomes by promoting functionality and actively involving the family throughout the process. Current studies show substantial heterogeneity in the outcomes selected for measurement, with missing or inadequate reporting of outcome definitions and limited reporting of the rationale for outcome selection. One solution to these issues is the development and implementation of a Core Outcome Set (COS) for children with CP undergoing physiotherapy interventions via telehealth. The objective of this study is to develop a COS for use in clinical research studies (i.e., clinical trials and observational studies) evaluating outcomes associated with physiotherapy interventions via telehealth in children with CP. This COS will be developed to be applicable to all children with cerebral palsy, regardless of their level of Gross Motor Function Classification System (GMFCS) classification.
Methods: The present study will consist of a study for the development of a Core Outcome Set (COS) to measure outcomes of physiotherapy interventions via telehealth for children with Cerebral Palsy (CP), which will be described following the Core Outcome Set–STAndards for Reporting: The COS-STAR Statement (KIRKHAM et al., 2016).
For the development of the COS, we will follow the recommendations established in Williamson et al., (2012): (1) a systematic review to generate a categorization list of potential outcomes for inclusion in the COS; (2) a consensus-building process involving key stakeholders involved in the care and/or research of children with CP (i.e., physiotherapists, researchers, and primary caregivers) to prioritize the outcomes for inclusion in the COS; and (3) a consensus meeting to refine and finalize the COS. A validation phase with a separate group of primary caregivers will also be completed.
Initially, a steering committee will be established, consisting of 10 experienced physiotherapists in the care of children with CP and researchers in this field. The committee will oversee all phases of the project through in-person meetings and video calls. Subsequently, two researchers will conduct a scoping review to identify the key outcomes and standardized instruments used to measure changes following telehealth physiotherapy interventions in children with CP. The review will follow the Preferred Reporting Items for Prism Extension for Scoping Reviews (PRISMA-ScR) guidelines.
The outcomes identified from the scoping review will form the basis for a survey to determine the outcomes used in telehealth physical therapy intervention studies for children with CP. Two stakeholder groups will be invited to complete the survey: physiotherapists and researchers, and parents of children with CP. The outcomes from these evaluators will be grouped and presented in the survey within central areas of a filter based from the International classification of Functioning, Disability and Health (ICF). The central areas to be included will be identified based on the impact of the health condition on the child, family, and healthcare system.
One group will consist of ten researchers, physicians, or experienced physiotherapists in studies involving children with CP from the multicenter research project PartiCipa Brasil. The other group will comprise three parents of children aged 0 to 12 years with CP who have undergone telehealth physiotherapy interventions. Participants will be asked to rate the importance of each outcome on a 5-point Likert scale (0=not important at all, 1=somewhat unimportant, 2=neutral, 3=important, 4=very important). Physiotherapists and researchers will be requested to rate all outcomes within all central areas. Only outcomes classified as "important" or "very important" by 85% or more of the respondents will be taken to the consensus meeting.
Primary caregivers will be asked to assess the outcomes in the main areas, including child-centered outcomes. A consensus meeting via video conference will be conducted to reach an agreement on the minimum set of outcomes that would be relevant to all stakeholders. The invited participants will include all members of the study's steering committee, physiotherapists, researchers, and parents who took part in the survey, as well as colleagues with substantial expertise in this research area. The outcomes will be presented according to the central areas identified through the scoping review conducted in Phase 1. Before the voting process, this approach will be explained to encourage voting within the context of the specific central area under focus.
The numerical outcomes of the survey (i.e., the proportion of physiotherapists and researchers who classified the outcome as "important" or "very important" and the frequency count for each Likert score option for primary caregivers) will be provided before each vote. The outcomes will be anonymously voted upon, with options for 'yes' (i.e., include in the final COS), 'no' (i.e., exclude from the final COS), and 'maybe' (i.e., reserve for further discussion). Outcomes for which 70% or more of the participants vote 'yes' will be considered as reaching a consensus for inclusion. If consensus is not reached, and if the total proportion of 'yes' and 'maybe' reaches or exceeds 70%, the outcome will be retained for further discussion. Any outcome that does not meet the criteria for consensus inclusion or further discussion will be eliminated.
After the voting process, the outcomes that had been reserved for further discussion will be re-examined. Participants will be given two options for each remaining outcome: merge it with an already included outcome or vote to include the outcome in the COS. Upon reaching a consensus on all items, the consensus group members will define the COS. The validation of the approved COS will be conducted with a second group of 12 parents of children with CP. In a 30 to 45-minute video conference session, the participating parents will be visually presented with the list of outcomes (List 1) in each of the ten main areas established in the approved COS, as well as the list of outcomes in each central area that were not included in the COS (List 2). All participants will be asked the following question: "In your experience, is any item from List 2 more important than the items from List 1?" If they respond 'yes', they will be invited to swap outcomes from List 1 with List 2. The outcomes from the COS will be considered valid for inclusion in the final COS (List 1) when at least two-thirds of the participants do not suggest swapping them for another outcome from List 2.