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

Between loss and restoration:The role of liminality in advancing theories of grief and bereavement

A recent national survey of bereaved partners found high levels of complicated grief and psychological distress, with evidence that loneliness and isolation may contribute to these outcomes. However, the mechanisms of action for this have not been explored. To advance grief theory this paper reports analysis of the survey free-text data to examine the relationship between social support and emotional responses to bereavement. Individuals bereaved of a civil partner or spouse 6-10 months previously were identified through death registration data. 569/1945 (29 %) completed surveys were received. Of those, 311 participants (55 %) provided responses to two free-text questions which asked about their 'feelings since the death of their partner or spouse', and 'about the support around' them. Data were analysed using corpus-assisted discourse analysis and the discourse dynamics approach for figurative language. Participants described diverse emotional responses to the bereavement (e.g. sadness, anger, denial, acceptance), and the value of formal and informal bereavement support. Although many of the experiences described are accounted for in existing grief theory, some participants described a liminal experience not recognised within these theories. They felt trapped, unable to engage with loss or restoration, and unable to move forward as their planned future no longer existed. They sought out 'communitas' (solidarity in experiences), but often found support from their social networks had diminished. Metaphors were used to describe this liminality, with partner grief expressed as a dark agentic force, a monster, an abyss, and as water. The findings of this study offer original insights into experiences and trajectories of bereavement, and our understandings of prolonged or complicated grief. A novel model 'Between Loss and Restoration' is presented to include these experiences. Recognition of the place for liminality within the spectrum of grief experiences could enhance grief literacy and improve formal and informal bereavement support provision.</p

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

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

Strategies to reduce waiting times in outpatient rehabilitation services for adults with physical disabilities : a systematic literature review

Objective: Identifying effective strategies to reduce waiting times is a crucial issue in many areas of health services. Long waiting times for rehabilitation services have been associated with numerous adverse effects in people with disabilities. The main objective of this study was to conduct a systematic literature review to assess the effectiveness of service redesign strategies to reduce waiting times in outpatient rehabilitation services for adults with physical disabilities. Methods: We conducted a systematic review, searching three databases (MEDLINE, CINAHL and EMBASE) from their inception until May 2021. We identified studies with comparative data evaluating the effect of rehabilitation services redesign strategies on reducing waiting times. The Mixed Methods Appraisal Tool was used to assess the methodological quality of the studies. A narrative synthesis was conducted. Results: Nineteen articles including various settings and populations met the selection criteria. They covered physiotherapy (n = 11), occupational therapy (n = 2), prosthetics (n = 1), exercise physiology (n = 1) and multidisciplinary (n = 4) services. The methodological quality varied (n = 10 high quality, n = 6 medium, n = 3 low); common flaws being missing information on the pre-redesign setting and characteristics of the populations. Seven articles assessed access processes or referral management strategies (e.g. self-referral), four focused on extending/modifying the roles of service providers (e.g. to triage) and eight changed the model of care delivery (e.g. mode of intervention). The different redesign strategies had positive effects on waiting times in outpatient rehabilitation services. Conclusions: This review highlights the positive effects of many service redesign strategies. These findings suggest that there are several effective strategies to choose from to reduce waiting times and help better respond to the needs of persons experiencing physical disabilities

The importance of integration of stakeholder views in core outcome set development: Otitis Media with Effusion in children with cleft palate

© 2015 Harman et al. This is an open access article distributed under the terms of the Creative Commons Attribution License, which permits unrestricted use, distribution, and reproduction in any medium, provided the original author and source are credited. Background: Approximately 75% of children with cleft palate (CP) have Otitis Media with Effusion (OME) histories. Evidence for the effective management of OME in these children is lacking. The inconsistency in outcome measurement in previous studies has led to a call for the development of a Core Outcome Set (COS). Despite the increase in the number of published COS, involvement of patients in the COS development process, and methods to integrate the views of patients and health professionals, to date have been limited. Methods and Findings: A list of outcomes measured in previous research was identified through reviewing the literature. Opinion on the importance of each of these outcomes was then sought from key stakeholders: Ear, Nose and Throat (ENT) surgeons, audiologists, cleft surgeons, speech and language therapists, specialist cleft nurses, psychologists, parents and children. The opinion of health professionals was sought in a three round Delphi survey where participants were asked to score each outcome using a bespoke online system. Parents and children were also asked to score outcomes in a survey and provided an in-depth insight into having OME through semi-structured interviews. The results of the Delphi survey, interviews and parent/patient survey were brought together in a final consensus meeting with representation from all stakeholders. A final set of eleven outcomes reached the definition of "consensus in" to form the recommended COS: hearing; chronic otitis media (COM); OME; receptive language skills; speech development; psycho social development; acute otitis media (AOM); cholesteatoma; side effects of treatment; listening skills; otalgia. Conclusions: We have produced a recommendation about the outcomes that should be measured, as a minimum, in studies of the management of OME in children with CP. The development process included input from key stakeholders and used novel methodology to integrate the opinion of healthcare professionals, parents and children

UK-born Pakistani-origin infants are relatively more adipose than white British infants: findings from 8704 mother-offspring pairs in the Born-in-Bradford prospective birth cohort

Background: Previous studies have shown markedly lower birth weight among infants of South Asian origin compared with those of White European origin. Whether such differences mask greater adiposity in South Asian infants and whether they persist across generations in contemporary UK populations is unclear. Our aim was to compare birth weight, skinfold thickness and cord leptin between Pakistani and White British infants and to investigate the explanatory factors, including parental and grandparental birthplace. Methods: We examined the differences in birth weight and skinfold thickness between 4649 Pakistani and 4055 White British infants born at term in the same UK maternity unit and compared cord leptin in a subgroup of 775 Pakistani and 612 White British infants. Results: Pakistani infants were lighter (adjusted mean difference −234 g 95% CI −258 to −210) and were smaller in both subscapular and triceps skinfold measurements. The differences for subscapular and triceps skinfold thickness (mean z-score difference −0.27 95% CI −0.34 to −0.20 and −0.23 95% CI −0.30 to −0.16, respectively) were smaller than the difference in birth weight (mean z-score difference −0.52 95% CI −0.58 to −0.47) and attenuated to the null with adjustment for birth weight (0.03 95% CI −0.03 to 0.09 and −0.01 95% CI −0.08 to 0.05, respectively). Cord leptin concentration (indicator of fat mass) was similar in Pakistani and White British infants without adjustment for birth weight, but with adjustment became 30% higher (95% CI 17% to 44%) among Pakistani infants compared with White British infants. The magnitudes of difference did not differ by generation. Conclusions: Despite being markedly lighter, Pakistani infants had similar skinfold thicknesses and greater total fat mass, as indicated by cord leptin, for a given birth weight than White British infants. Any efforts to reduce ethnic inequalities in birth weight need to consider differences in adiposity and the possibility that increasing birth weight in South Asian infants might inadvertently worsen health by increasing relative adiposity