Design and Administration of Patient-Centred Outcome Measures: The Perspectives of Children and Young People with Life-Limiting or Life-Threatening Conditions and Their Family Members

Abstract

Background Self-reported health data from children with life-limiting conditions is rarely collected. To improve acceptability and feasibility of child and family-centred outcome measures for children, they need to be designed in a way that reflects preferences, priorities and abilities. Objectives The aim was to identify preferences for patient-reported outcome measure design (recall period, response format, length, administration mode) to improve the feasibility, acceptability, comprehensibility and relevance of a child and family-centred outcome measure, among children with life-limiting conditions and their family members. Method A semi-structured qualitative interview study seeking the perspectives of children with life-limiting conditions, their siblings and parents on measure design was conducted. Participants were purposively sampled and recruited from nine UK sites. Verbatim transcripts were analysed using framework analysis. Results A total of 79 participants were recruited: 39 children aged 5–17 years (26 living with a life-limiting condition; 13 healthy siblings) and 40 parents (of children aged 0–17 years). Children found a short recall period and a visually appealing measure with ten questions or fewer most acceptable. Children with life-limiting conditions were more familiar with using rating scales such as numeric and Likert than their healthy siblings. Children emphasised the importance of completing the measure alongside interactions with a healthcare professional to enable them to talk about their responses. While parents assumed that electronic completion methods would be most feasible and acceptable, a small number of children preferred paper. Conclusions This study demonstrates that children with life-limiting conditions can engage in communicating preferences regarding the design of a patient-centred outcome measure. Where possible, children should be given the opportunity to participate in the measure development process to enhance acceptability and uptake in clinical practice. Results of this study should be considered in future research on outcome measure development in children.

What are the anticipated benefits, risks, barriers and facilitators to implementing person-centred outcome measures into routine care for children and young people with life-limiting and life-threatening conditions? A qualitative interview study with key stakeholders

“… 74 , 75 Nevertheless, it is important to ensure that PCOMs are used in a meaningful way whereby PCOMs actively support children to participate in their own care and shared decision-making and to avoid it becoming tokenistic whereby children are just asked to complete a PCOM but with no additional discission, involvement or feedback. 6 Given the expressed preference by children for measure completion to be integrated within conversations with their healthcare team, 49 there may be impacts to clinical workflow beyond those identified in the adult care situation.…”

“…This cross-sectional qualitative interview study 47 is reported in accordance with the consolidated criteria for reporting qualitative studies (COREQ). 55 It sits within a sequential mixed-methods study to develop, 35 , 47 – 49 , 56 – 59 validate 51 and implement 60 – 62 a novel PCOM for children with any life-limiting illness. Qualitative interviews were conducted during the development phase with the aims of identifying priority items to include in the measure, 47 preferences for design and administration modes, 49 and potential benefits and challenges of implementing a new PCOM into routine care.…”

“… 55 It sits within a sequential mixed-methods study to develop, 35 , 47 – 49 , 56 – 59 validate 51 and implement 60 – 62 a novel PCOM for children with any life-limiting illness. Qualitative interviews were conducted during the development phase with the aims of identifying priority items to include in the measure, 47 preferences for design and administration modes, 49 and potential benefits and challenges of implementing a new PCOM into routine care. The data related to implementation collected during the development phase are reported here and will inform the implementation phase.…”

“…The concept of information power or pragmatic saturation 67 was used to determine the required sample size to address all of the aims of the original study. 47 , 49 , 60 , 61 Due to the range of aims being met and the heterogeneity of the population, the dataset required diversity and depth.…”

“…The CPOS:UK (Children’s Palliative care Outcome Scale: UK version) study aims to develop, validate and implement a novel PCOM for all children with any life-limiting condition in the UK. Five initial versions of the C-POS:UK have been developed to reflect variation in age/developmental stages of the target population and allow for proxy reporting if required 47 – 50 following the Consensus-based Standards for the selection of health Measurement Instruments (COSMIN) 52 , 53 and Rothrock guidance. 54 However, future implementation to be successful, implementation strategies must be informed by the views and preferences of key stakeholders: children with life-limiting conditions, their family members and professionals involved in their care.…”