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

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

‘My life is a mess but I cope’: An analysis of the language children and young people use to describe their own life-limiting or life-threatening condition

Background: Children and young people with life-limiting and life-threatening conditions have multidimensional needs and heterogenous cognitive and communicative abilities. There is limited evidence to support clinicians to tailor their communication to each individual child. Aim: To explore the language children and young people use to describe their own condition, to inform strategies for discussing needs and priorities. Design: Positioned within a social constructivist paradigm, a secondary discourse analysis of semi-structured interview data was conducted incorporating the discourse dynamics approach for figurative language. Setting/participants: A total of 26 children and young people aged 5–17 years with life-limiting or life-threatening conditions (6 cancer; 20 non-cancer) were recruited from nine clinical services (six hospitals and three hospices) across two UK nations. Results: The language children and young people use positions them as ‘experts in their condition’. They combine medical terminology with their preferred terms for their body to describe symptoms and treatments, and use comparatives and superlatives to communicate their health status. Their language depicts their condition as a ‘series of (functional and social) losses’, which single them out from their peers as ‘the sick one’. Older children and young people also incorporate figurative language to expand their descriptions. Conclusion/discussion: Children and young people can provide rich descriptions of their condition. Paying attention to their lexical choices, and converging one’s language towards theirs, may enable more child-centred discussions. Expanding discussions about ‘what matters most’ with consideration of the losses and differences they have experienced may facilitate a fuller assessment of their concerns, preferences and priorities

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

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

Spiritual, religious, and existential concerns of children and young people with life-limiting and life-threatening conditions: A qualitative interview study

BACKGROUND: Despite being a core domain of palliative care, primary data on spiritual and existential concerns has rarely been collected among children with life-limiting and life-threatening conditions and their families. Existing evidence has tended to focus on the religious aspects among children with cancer. AIM: To identify the spiritual needs of children with life-limiting and life-threatening conditions. DESIGN: Cross-sectional semi-structured, qualitative interview study with children, families and health and social care professionals. Verbatim transcripts were analysed using Framework analysis. SETTING/PARTICIPANTS: Purposively sampled children with life-limiting and life-threatening conditions, their parents and siblings, health and social care professionals recruited from six hospitals and three children's hospices in the UK, and commissioners of paediatric palliative care services recruited through networks and a national charity. RESULTS: One hundred six participants were interviewed: 26 children (5-17 years), 53 family members (parents/carers of children 0-17 years and siblings (5-17 years)), 27 professionals (health and social care professionals and commissioners of paediatric palliative care). Themes included: living life to the fullest, meaning of life and leaving a legacy, uncertainty about the future, determination to survive, accepting or fighting the future and role of religion. Children as young as 5 years old identified needs or concerns in the spiritual domain of care. CONCLUSIONS: Addressing spiritual concerns is essential to providing child- and family-centred palliative care. Eliciting spiritual concerns may enable health and social care professionals to identify the things that can support and enhance a meaningful life and legacy for children and their families

Spiritual, religious, and existential concerns of children and young people with life-limiting and life-threatening conditions: A qualitative interview study

Background:Despite being a core domain of palliative care, primary data on spiritual and existential concerns has rarely been collected among children with life-limiting and life-threatening conditions and their families. Existing evidence has tended to focus on the religious aspects among children with cancer.Aim:To identify the spiritual needs of children with life-limiting and life-threatening conditions.Design:Cross-sectional semi-structured, qualitative interview study with children, families and health and social care professionals. Verbatim transcripts were analysed using Framework analysisSetting/participants:Purposively sampled children with life-limiting and life-threatening conditions, their parents and siblings, health and social care professionals recruited from six hospitals and three children’s hospices in the UK, and commissioners of paediatric palliative care services recruited through networks and a national charity.Results:One hundred six participants were interviewed: 26 children (5–17 years), 53 family members (parents/carers of children 0–17 years and siblings (5–17 years)), 27 professionals (health and social care professionals and commissioners of paediatric palliative care). Themes included: living life to the fullest, meaning of life and leaving a legacy, uncertainty about the future, determination to survive, accepting or fighting the future and role of religion. Children as young as 5 years old identified needs or concerns in the spiritual domain of care.Conclusions:Addressing spiritual concerns is essential to providing child- and family-centred palliative care. Eliciting spiritual concerns may enable health and social care professionals to identify the things that can support and enhance a meaningful life and legacy for children and their families

Implementation of child-centred outcome measures in routine paediatric healthcare practice: a systematic review

Background: Person-centred outcome measures (PCOMs) are commonly used in routine adult healthcare to measure and improve outcomes, but less attention has been paid to PCOMs in children’s services. The aim of this systematic review is to identify and synthesise existing evidence of the determinants, strategies, and mechanisms that influence the implementation of PCOMs into paediatric healthcare practice. Methods: The review was conducted and reported in accordance with PRISMA guidelines. Databased searched included CINAHL, Embase, Medline, and PsycInfo. Google scholar was also searched for grey literature on 25th March 2022. Studies were included if the setting was a children’s healthcare service, investigating the implementation or use of an outcome measure or screening tool in healthcare practice, and reported outcomes relating to use of a measure. Data were tabulated and thematically analysed through deductive coding to the constructs of the adapted-Consolidated Framework for Implementation Research (CFIR). Results were presented as a narrative synthesis, and a logic model developed. Results: We retained 69 studies, conducted across primary (n = 14), secondary (n = 13), tertiary (n = 37), and community (n = 8) healthcare settings, including both child self-report (n = 46) and parent-proxy (n = 47) measures. The most frequently reported barriers to measure implementation included staff lack of knowledge about how the measure may improve care and outcomes; the complexity of using and implementing the measure; and a lack of resources to support implementation and its continued use including funding and staff. The most frequently reported facilitators of implementation and continued use include educating and training staff and families on: how to implement and use the measure; the advantages of using PCOMs over current practice; and the benefit their use has on patient care and outcomes. The resulting logic model presents the mechanisms through which strategies can reduce the barriers to implementation and support the use of PCOMs in practice. Conclusions: These findings can be used to support the development of context-specific implementation plans through a combination of existing strategies. This will enable the implementation of PCOMs into routine paediatric healthcare practice to empower settings to better identify and improve child-centred outcomes. Trial registration: Prospero CRD 42022330013

Próffræðilegir eiginleikar íslenskrar þýðingar GAD-7 í klínísku úrtaki

GAD-7 er sjö atriða sjálfsmatslisti sem ætlað er að skima fyrir almennri kvíðaröskun og meta alvarleika kvíðaeinkenna. Erlendis hefur verið sýnt fram á góðan innri áreiðanleika listans en þáttabygging hans hefur reynst óstöðug og sértækni léleg. Markmið rannsóknarinnar var að kanna próffræðilega eiginleika íslenskrar þýðingar GAD-7 í fyrsta skipti. Var innri áreiðanleiki og þáttaformgerð skoðuð í klínísku úrtaki með tölfræðiaðgerðum sem henta gögnum sem mæld eru á raðkvarða. Niðurstöður sýndu fram á góðan innri áreiðanleika mælinganna en lágt meðaltal heildarskora og lágt hlutfall skimunar í áhættuhóp miðað við tegund úrtaks. Auk þess kom fram skýr eins þáttar formgerð sem styður við réttmæti listans. Þáttagreining sýndi fram á framúrskarandi niðurstöðu fyrstu fjögurra atriða listans en niðurstöður voru áberandi verri fyrir síðustu þrjú atriðin. Bendir þetta til þess að seinni atriðin virki ekki sem skyldi í íslenskri þýðingu GAD-7. Þessi atriði voru skoðuð nánar og lagðar fram tillögur um mögulegar lagfæringar á þeim, en rannsóknin sýnir að íslensk þýðing listans krefst frekari rannsókna og úrbóta. Auk þess var sýnt fram á mikilvægi þess að kanna próffræðilega eiginleika þýddra mælitækja áður en þau eru tekin í notkun