Achieving consensus on priority items for paediatric palliative care outcome measurement: Results from a modified Delphi survey, engagement with a children’s research involvement group and expert item generation

Abstract

Background: There is no validated outcome measure for use in children’s palliative care outside sub-Saharan Africa. Stakeholders must be involved in the development of such measures to ensure face and content validity. Aim: To gain expert stakeholder consensus on items for inclusion in a paediatric palliative care outcome measure to establish face and content validity. Design: This study was conducted in two phases following Rothrock and COSMIN guidance on patient-reported outcome measure development. Phase 1: Three-round modified Delphi survey to establish consensus on priority items. Phase 2: Item generation meeting with key stakeholders to develop initial measure versions. A young person’s advisory group was also consulted on priority outcomes. Setting and participants: Delphi survey: Parents and professionals with experience of caring for a child with a life-limiting condition. Young person’s advisory group: young people age 10–20 years. Item generation meeting: bereaved parents, academics and clinicians. Results: Phase 1: Delphi survey ( n = 82). Agreement increased from Kendall’s W = 0.17 to W = 0.61, indicating movement towards consensus. Agreement between professional and parent ranking was poor (Cohen’s kappa 0.13). Professionals prioritised physical symptoms, whereas parents prioritised psychosocial and practical concerns. Advisory group: Children ( n = 22) prioritised items related to living a ‘normal life’ in addition to items prioritised by adult participants. Phase 2: Five age/developmental stage appropriate child and proxy-reported versions of C-POS, containing 13 items, were drafted. Conclusions: This study highlights the importance and feasibility of involving key stakeholders in PROM item generation, as important differences were found in the priority outcomes identified by children, parents and professionals.

Cognitive testing of the Children’s Palliative Outcome Scale (C-POS) with children, young people and their parents/carers

“…29 Previous studies that have shown that children with life-limiting conditions prefer to complete a measure in the presence of an adult in case they have questions or need support. 12 We found some instances of social desirability bias in children 5-7 years (or cognitive equivalent) who gave the answers they thought the researcher wanted to hear (e.g. saying they had not experienced any pain when in fact they had).…”

“…Children <8-years-old tested a 3-point response format and recall of yesterday and today. The use of emojis to anchor responses was informed by evidence from previous work 12 with a smiling emoji representing the positive outcome for the item. All child versions of C-POS had eight questions.…”

“…[6][7][8] Item generation and content validity were informed by a qualitative semi-structured interview study, 9,10 a systematic review to inform optimal recall period, response format and administration mode, 11 a Delphi survey to achieve consensus on priority outcomes and an item generation meeting. 12 This has resulted in five versions of C-POS -two parent/proxy versions (child <2 years and ⩾2 years) and three child versions (5-7, 8-12 and 13-17 years or cognitive equivalent). The concepts explored are the same in each version, with simpler language for younger/less cognitively able children (Table 1).…”

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

“…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][48][49][56][57][58][59] validate 51 and implement [60][61][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.…”

“…[41][42][43][44][45][46] 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 lifelimiting 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][48][49][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.…”