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

BACKGROUND: The Children's Palliative Outcome Scale (C-POS) is being developed using best methodological guidance on outcome measure development, This recommends cognitive testing, an established method of item improvement, prior to psychometric testing. AIM: To cognitively test C-POS within the target population to establish comprehensibility, comprehensiveness, relevance and acceptability. DESIGN: Cross-sectional cognitive interview study following COnsensus-based Standards for the selection of health Measurement INstruments (COSMIN) methodology and Rothrock guidance on outcome measure development. Cognitive interviews were conducted using 'think aloud' and verbal probing techniques. SETTING/PARTICIPANTS: Children 5-⩽17 years old with life-limiting conditions and parents/carers of children with life-limiting conditions were recruited from 14 UK sites. RESULTS: Forty-eight individuals participated (36 parents; 12 children) in cognitively testing the five versions of C-POS over two to seven rounds. Content and length were acceptable, and all questions were considered important. Refinements were made to parent/carer versions to be inclusive of non-verbal children such as changing 'share' to 'express' feelings; and 'being able to ask questions' to 'having the appropriate information'. Changes to improve comprehensibility of items such as 'living life to the fullest' were also made. Parents reported that completing an outcome measure can be distressing but this is anticipated and that being asked is important. CONCLUSION: Cognitive interviewing has facilitated refinement of the C-POS, especially for non-verbal children who represent a large proportion of those with a life-limiting condition. This study has enhanced the face and content validity of the measure and provided preliminary evidence for acceptability for use in routine practice.

Psychosocial support interventions for children and adolescents experiencing gender dysphoria or incongruence: a systematic review.

BACKGROUND: National and international guidelines recommend that psychosocial support should be a key component of the care offered to children and adolescents experiencing gender dysphoria/incongruence. However, specific approaches or interventions are not recommended. AIM: To identify and summarise evidence on the outcomes of psychosocial support interventions for children and adolescents (age 0-18) experiencing gender dysphoria/incongruence. METHODS: Systematic review and narrative synthesis. Database searches (MEDLINE; EMBASE; CINAHL; PsycINFO; Web of Science) were performed in April 2022, with results assessed independently by two reviewers. Peer-reviewed articles reporting the results of studies measuring outcomes of psychosocial support interventions were included. Quality was assessed using the Mixed Methods Appraisal Tool. RESULTS: Ten studies were included. Half were conducted in the US, with others from Australia, Canada, New Zealand and the UK. Six were pre-post analyses or cohort studies, three were mixed methods, and one was a secondary analysis of intervention data from four trials. Most studies were of low quality. Most analyses of mental health and psychosocial outcomes showed either benefit or no change, with none indicating negative or adverse effects. CONCLUSIONS: The small number of low-quality studies limits conclusions about the effectiveness of psychosocial interventions for children/adolescents experiencing gender dysphoria/incongruence. Clarity on the intervention approach as well as the core outcomes would support the future aggregation of evidence. More robust methodology and reporting is required. PROSPERO REGISTRATION NUMBER: CRD42021289659.

Characteristics of children and adolescents referred to specialist gender services: a systematic review.

BACKGROUND: Increasing numbers of children/adolescents experiencing gender dysphoria/incongruence are being referred to specialist gender services. Services and practice guidelines are responding to these changes. AIM: This systematic review examines the numbers and characteristics of children/adolescents (under 18) referred to specialist gender or endocrinology services. METHODS: Database searches were performed (April 2022), with results assessed independently by two reviewers. Peer-reviewed articles providing at least birth-registered sex or age at referral were included. Demographic, gender-related, mental health, neurodevelopmental conditions and adverse childhood experience data were extracted. A narrative approach to synthesis was used and where appropriate proportions were combined in a meta-analysis. RESULTS: 143 studies from 131 articles across 17 countries were included. There was a twofold to threefold increase in the number of referrals and a steady increase in birth-registered females being referred. There is inconsistent collection and reporting of key data across many of the studies. Approximately 60% of children/adolescents referred to services had made steps to present themselves in their preferred gender. Just under 50% of studies reported data on depression and/or anxiety and under 20% reported data on other mental health issues and neurodevelopmental conditions. Changes in the characteristics of referrals over time were generally not reported. CONCLUSIONS: Services need to capture, assess and respond to the potentially co-occurring complexities of children/adolescents being referred to specialist gender and endocrine services. Agreement on the core characteristics for collection at referral/assessment would help to ensure services are capturing data as well as developing pathways to meet the needs of these children.PROSPERO registration number CRD42021289659.

Interventions to suppress puberty in adolescents experiencing gender dysphoria or incongruence: a systematic review.

BACKGROUND: Treatment to suppress or lessen effects of puberty are outlined in clinical guidelines for adolescents experiencing gender dysphoria/incongruence. Robust evidence concerning risks and benefits is lacking and there is a need to aggregate evidence as new studies are published. AIM: To identify and synthesise studies assessing the outcomes of puberty suppression in adolescents experiencing gender dysphoria/incongruence. METHODS: A systematic review and narrative synthesis. Database searches (Medline, Embase, CINAHL, PsycINFO, Web of Science) were performed in April 2022, with results assessed independently by two reviewers. An adapted version of the Newcastle-Ottawa Scale for cohort studies was used to appraise study quality. Only moderate-quality and high-quality studies were synthesised. The Preferred Reporting Items for Systematic Reviews and Meta-Analyses reporting guidelines were used. RESULTS: 11 cohort, 8 cross-sectional and 31 pre-post studies were included (n=50). One cross-sectional study was high quality, 25 studies were moderate quality (including 5 cohort studies) and 24 were low quality. Synthesis of moderate-quality and high-quality studies showed consistent evidence demonstrating efficacy for suppressing puberty. Height increased in multiple studies, although not in line with expected growth. Multiple studies reported reductions in bone density during treatment. Limited and/or inconsistent evidence was found in relation to gender dysphoria, psychological and psychosocial health, body satisfaction, cardiometabolic risk, cognitive development and fertility. CONCLUSIONS: There is a lack of high-quality research assessing puberty suppression in adolescents experiencing gender dysphoria/incongruence. No conclusions can be drawn about the impact on gender dysphoria, mental and psychosocial health or cognitive development. Bone health and height may be compromised during treatment. More recent studies published since April 2022 until January 2024 also support the conclusions of this review. PROSPERO REGISTRATION NUMBER: CRD42021289659.

Clinical guidelines for children and adolescents experiencing gender dysphoria or incongruence: a systematic review of guideline quality (part 1).

BACKGROUND: Increasing numbers of children and adolescents experiencing gender dysphoria/incongruence are being referred to specialist gender services. There are various guidelines outlining approaches to the clinical care of these children and adolescents. AIM: To examine the quality and development of published guidelines or clinical guidance containing recommendations for managing gender dysphoria/incongruence in children and/or adolescents (age 0-18). A separate paper reports the synthesis of guideline recommendations. METHODS: A systematic review and narrative synthesis. Databases (Medline, Embase, CINAHL, PsycINFO, Web of Science) were searched to April 2022 and web-based searches and contact with international experts continued to December 2022, with results assessed independently by two reviewers. The Appraisal of Guidelines for Research and Evaluation tool was used to examine guideline quality. RESULTS: Twenty-three guidelines/clinical guidance publications (1998-2022) were identified (4 international, 3 regional and 16 national). The quality and methods reporting in these varied considerably. Few guidelines systematically reviewed empirical evidence, and links between evidence and recommendations were often unclear. Although most consulted with relevant stakeholders, including 10 which involved service users or user representatives, it was often unclear how this influenced recommendations and only two reported including children/adolescents and/or parents. Guidelines also lacked clarity about implementation. Two international guidelines (World Professional Association for Transgender Health and Endocrine Society) formed the basis for most other guidance, influencing their development and recommendations. CONCLUSIONS: Most clinical guidance for managing children/adolescents experiencing gender dysphoria/incongruence lacks an independent and evidence-based approach and information about how recommendations were developed. This should be considered when using these to inform service development and clinical practice. PROSPERO REGISTRATION NUMBER: CRD42021289659.

Gender services for children and adolescents across the EU-15+ countries: an online survey.

BACKGROUND: Over the last 10-15 years, there has been an increase in the number of children and adolescents referred to gender services, particularly among adolescent birth-registered females. This population shows a higher prevalence of co-occurring mental health difficulties and neurodevelopmental conditions. Some countries have recently restricted access to medical treatments in recognition of the uncertain evidence base. AIM: To understand the current provision of gender services for children and adolescents across the EU-15+ countries that have comparable high-income healthcare systems, to inform service development in the UK. METHODS: An e-survey of paediatric gender services was conducted between September 2022 and April 2023. It covered service structure, care pathways, interventions and data collection. Data were described and compared to identify similarities and differences among participating services. RESULTS: 15 services in eight countries (Australia, Belgium, Denmark, Norway, Northern Ireland, The Netherlands, Spain and Finland) responded. While a multidisciplinary team was present in all services, its composition and organisation varied. Clinical practice was informed by international guidelines, with four countries following their own national guidelines. Differences were observed in referral criteria, care pathways for prepubertal children and those with co-occurring conditions. Eligibility criteria for medical interventions also varied. Psychosocial support and interventions were limited, and outcome data collection was scarce. CONCLUSIONS: This survey revealed both similarities and key variations in the clinical practice of paediatric gender services across eight different countries. The study emphasises the need for service development that both considers the management of co-occurring conditions and embeds routine data collection in practice.

Care pathways of children and adolescents referred to specialist gender services: a systematic review.

BACKGROUND: Increasing numbers of children and adolescents experiencing gender dysphoria/incongruence are being referred to specialist gender services. However, little is currently known about the proportions accessing different types of care and treatment following referral. AIM: This systematic review examines the range of care pathways of children/adolescents (under 18) referred to specialist gender or endocrinology services. METHODS: Database searches were performed (April 2022), with results assessed independently by two reviewers. Peer-reviewed articles providing data for numbers of children and/or adolescents at referral/assessment and their treatment pathways were included. A narrative approach to synthesis was used and where appropriate proportions were combined in a random-effects meta-analysis. RESULTS: 23 studies across nine countries were included, representing 6133 children and/or adolescents with a median age at assessment of 14-16 and overall a higher percentage of birth-registered females. Of those assessed, 36% (95% CI 27% to 45%) received puberty suppression, 51% (95% CI 40% to 62%) received masculinising or feminising hormones, 68% (95% CI 57% to 77%) received puberty suppression and/or hormones and 16% (95% CI 10% to 24%) received surgery. No study systematically reported information about the full pathway or psychological care received by children/adolescents. Follow-up in many studies was insufficient or unclear. Reasons for discontinuation were rarely provided. CONCLUSIONS: Prospective studies with long-term follow-up reporting information about the full range of pathways are needed to understand what happens to children and adolescents referred to specialist gender services. Information about provision of psychological care is needed considering high rates of psychosocial difficulties in this population.PROSPERO registration number CRD42021289659.

Clinical guidelines for children and adolescents experiencing gender dysphoria or incongruence: a systematic review of recommendations (part 2).

BACKGROUND: Increasing numbers of children and adolescents experiencing gender dysphoria/incongruence are being referred to specialist gender services and there are various published guidelines outlining approaches to clinical care. AIM: To examine the recommendations about the management of children and/or adolescents (age 0-18) experiencing gender dysphoria/incongruence in published guidelines or clinical guidance. A separate paper examines the quality and development of guidelines. METHODS: A systematic review and narrative synthesis. Databases (Medline, Embase, CINAHL, PsycINFO, Web of Science) were searched to April 2022 and web-based searches and contact with international experts continued to December 2022, with results assessed independently by two reviewers. The Appraisal of Guidelines for Research and Evaluation tool was used to examine guideline quality. RESULTS: 23 guidelines/clinical guidance publications (1998-2022) were identified (4 international, 3 regional, 16 national). Guidelines describe a similar care pathway starting with psychosocial care for prepubertal children, puberty suppressants followed by hormones for eligible adolescents and surgical interventions as these adolescents enter adulthood. In general, there is consensus that adolescents should receive a multidisciplinary assessment, although clear guidance about the purpose or approach is lacking. There are differing recommendations about when and on what basis psychological and medical interventions should be offered. There is limited guidance about what psychological care should be provided, about the management of prepubertal children or those with a non-binary gender identity, nor about pathways between specialist gender services and other providers. CONCLUSIONS: Published guidance describes a similar care pathway; however, there is no current consensus about the purpose and process of assessment for children or adolescents with gender dysphoria/incongruence, or about when psychological or hormonal interventions should be offered and on what basis. PROSPERO REGISTRATION NUMBER: CRD42021289659.

Masculinising and feminising hormone interventions for adolescents experiencing gender dysphoria or incongruence: a systematic review.

BACKGROUND: Clinical guidelines outline the use of hormones for masculinisation/feminisation in adolescents experiencing gender dysphoria or incongruence. Robust evidence concerning risks and benefits is lacking. There is a need to aggregate evidence as research becomes available. AIM: Identify and synthesise studies assessing the outcomes of hormones for masculinisation/feminisation in adolescents experiencing gender dysphoria/incongruence. METHODS: Systematic review and narrative synthesis. Database searches (MEDLINE, Embase, CINAHL, PsycINFO, Web of Science) were performed in April 2022, with results assessed independently by two reviewers. An adapted version of the Newcastle-Ottawa Scale for Cohort Studies was used to assess study quality. Moderate- and high-quality studies were synthesised. RESULTS: 12 cohort, 9 cross-sectional and 32 pre-post studies were included (n=53). One cohort study was high-quality. Other studies were moderate (n=33) and low-quality (n=19). Synthesis of high and moderate-quality studies showed consistent evidence demonstrating induction of puberty, although with varying feminising/masculinising effects. There was limited evidence regarding gender dysphoria, body satisfaction, psychosocial and cognitive outcomes, and fertility. Evidence from mainly pre-post studies with 12-month follow-up showed improvements in psychological outcomes. Inconsistent results were observed for height/growth, bone health and cardiometabolic effects. Most studies included adolescents who received puberty suppression, making it difficult to determine the effects of hormones alone. CONCLUSIONS: There is a lack of high-quality research assessing the use of hormones in adolescents experiencing gender dysphoria/incongruence. Moderate-quality evidence suggests mental health may be improved during treatment, but robust study is still required. For other outcomes, no conclusions can be drawn. More recent studies published since April 2022 until January 2024 also support the conclusions of this review.PROSPERO registration number: CRD42021289659.

Impact of social transition in relation to gender for children and adolescents: a systematic review.

BACKGROUND: Increasing numbers of children and adolescents experiencing gender dysphoria or incongruence are being referred to specialist gender services. Historically, social transitioning prior to assessment was rare but it is becoming more common. AIM: To identify and synthesise studies assessing the outcomes of social transition for children and adolescents (under 18) experiencing gender dysphoria/incongruence. METHODS: A systematic review and narrative sythesis. Database searches (Medline, Embase, CINAHL, PsycINFO, Web of Science) were perfomed in April 2022. Studies reporting any outcome of social transition (full or partial) for children and adolescents experiencing gender dysphoria/incongruence were included. An adapted version of the Newcastle-Ottawa Scale for cohort studies was used to appraise study quality. RESULTS: Eleven studies were included (children (n=8) and adolescents (n=3)) and most were of low quality. The majority were from the US, featured community samples and cross-sectional analyses. Different comparator groups were used, and outcomes related to mental health and gender identity reported. Overall studies consistently reported no difference in mental health outcomes for children who socially transitioned across all comparators. Studies found mixed evidence for adolescents who socially transitioned. CONCLUSIONS: It is difficult to assess the impact of social transition on children/adolescents due to the small volume and low quality of research in this area. Importantly, there are no prospective longitudinal studies with appropriate comparator groups assessing the impact of social transition on mental health or gender-related outcomes for children/adolescents. Professionals working in the area of gender identity and those seeking support should be aware of the absence of robust evidence of the benefits or harms of social transition for children and adolescents. PROSPERO REGISTRATION NUMBER: CRD42021289659.

Inequalities in emergency care use across transition from paediatric to adult care: a retrospective cohort study of young people with chronic kidney disease in England.

Transition of young people with chronic kidney disease (CKD) from paediatric to adult healthcare has been associated with poor outcomes, but few population-level studies examine trends in subgroups. We aimed to assess sociodemographic inequalities in changes in unplanned secondary care utilisation occurring across transfer to adult care for people with CKD in England. A cohort was constructed from routine healthcare administrative data in England of young people with childhood-diagnosed CKD who transitioned to adult care. The primary outcome was the number of emergency inpatient admissions and accident and emergency department (A&E) attendances per person year, compared before and after transfer. Injury-related and maternity admissions were excluded. Outcomes were compared via sociodemographic data using negative binomial regression with random effects. The cohort included 4505 individuals. Controlling for age, birth year, age at transfer, region and sociodemographic factors, transfer was associated with a significant decrease in emergency admissions (IRR 0.75, 95% CI 0.64-0.88) and no significant change in A&E attendances (IRR 1.10, 95% CI 0.95-1.27). Female sex was associated with static admissions and increased A&E attendances with transfer, with higher admissions and A&E attendances compared to males pre-transfer. Non-white ethnicities and higher deprivation were associated with higher unplanned secondary care use. CONCLUSION:  Sociodemographic inequalities in emergency secondary care usage were evident in this cohort across the transition period, independent of age, with some variation between admissions and A&E use, and evidence of effect modification by transfer. Such inequalities likely have multifactorial origin, but importantly, could represent differential meetings of care needs. WHAT IS KNOWN: • In chronic kidney disease (CKD), transfer from paediatric to adult healthcare is associated with declining health outcomes. • Known differences in CKD outcomes by sociodemographic factors have limited prior exploration in the context of transfer. WHAT IS NEW: • Population-level data was used to examine the impacts of transfer and sociodemographic factors on unplanned secondary care utilisation in CKD. • Healthcare utilisation trends may not reflect known CKD pathophysiology and there may be unexplored sociodemographic inequalities in the experiences of young people across transfer.

'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.

Experiences of music therapy in paediatric palliative care from multiple stakeholder perspectives: A systematic review and qualitative evidence synthesis.

BACKGROUND: Children and young people with life-limiting conditions and their families need physical and emotional support to manage the challenges of their lives. There is a lack of synthesised qualitative research about how music therapy is experienced by children, young people and their families supported by paediatric palliative care services. AIM: To systematically identify and synthesise qualitative research on experiences of music therapy in paediatric palliative care from stakeholder perspectives. DESIGN: A Qualitative Evidence Synthesis was conducted using Thematic Synthesis. The review protocol was registered in PROSPERO (registration number: CRD42021251025). DATA SOURCES: Searches were conducted with no dates imposed via the electronic databases PsycINFO, MEDLINE, EMBASE, AMED and CINAHL in April 2021 and updated in April 2022. Studies were appraised for quality using the Critical Appraisal Skills Programme tool (CASP). RESULTS: A total of 148 studies were found, 5 studies met the eligibility criteria reporting the experiences of 14 mothers, 24 family members and 4 staff members in paediatric palliative care. There were five overarching themes: emotional and physical reprieve, opportunity for normalised experiences, thriving despite life limited condition, enhance family wellbeing and therapeutic relationship central to outcomes. CONCLUSION: Music therapy provides unique benefits for this paediatric population particularly in supporting child and family wellbeing. The therapeutic relationship, interpersonal skills of the therapist and experience in paediatric palliative care are perceived as central to these positive outcomes.

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.

BACKGROUND: There is a growing evidence-base underpinning implementation of person-centred outcome measures into adult palliative care. However evidence on how best to achieve this with children facing life-threatening and life-limiting conditions is limited. AIM: To identify the anticipated benefits, risks, barriers and facilitators to implementing person-centred outcome measures for children with life-limiting and life-threatening conditions. DESIGN: Cross-sectional qualitative semi-structured interview study with key stakeholders analysed using Framework analysis informed by the adapted-Consolidated Framework for Implementation Research. SETTING/PARTICIPANTS: A total of n = 26 children with life-limiting or life-threatening conditions, n = 40 parents/carers, n = 13 siblings and n = 15 health and social care professionals recruited from six hospitals and three children's hospices and n = 12 Commissioners of health services. RESULTS: All participants were supportive of future implementation of person-centred outcome measures into care. Anticipated benefits included: better understanding of patient and family priorities, improved communication and collaborative working between professionals and families and standardisation in data collection and reporting. Anticipated risks included increased workload for staff and measures not being used as intended. Implementation barriers included: acceptability and usability of outcome measures by children; burden and capacity of parents/carers regarding completion; privacy concerns; and language barriers. Implementation facilitators included designing measures using language that is meaningful to children and families, ensuring potential benefits of person-centred outcome measures are communicated to encourage 'buy-in' and administering measures with known and trusted professional. CONCLUSIONS: Implementation of person-centred outcome measures offer potential benefits for children with life-limiting and life-threatening conditions. Eight recommendations are made to maximise benefits and minimise risks in implementation.

Staff well-being in UK children's hospices: a national survey.

OBJECTIVES: Poor psychological well-being among healthcare workers can have numerous negative impacts, but evidence about levels of burnout in children's hospice care staff is limited. This study aimed to determine the prevalence of burnout and to explore the association between staff characteristics and support mechanisms with burnout among children's hospice care staff in the UK. METHODS: Two national online surveys collecting data on hospice care staff psychological well-being and hospice organisational characteristics. All children's hospices in the UK were invited.Thirty-one hospices (out of 52) responded to the hospice survey and 583 staff responded to the staff survey. Data collection took place between May and December 2020 and measures included the Copenhagen Burnout Inventory, Work Engagement and the Health and Safety Executive Management Standards Indicator Tool. RESULTS: Burnout prevalence was 11% and mean burnout score was 32.5 (SD: 13.1). Burnout levels were independent of working arrangements (eg, working from home or at the hospice) during the COVID-19 pandemic. Hospices performed well in most management standards, but poorly on the 'Control' domain. The average Work Engagement score for staff was 7.5 (SD: 1.5). CONCLUSIONS: Burnout levels for staff in children's hospices in the UK were lower than in other healthcare settings, with this comparing to 17.3% among palliative care staff generally. Overall, hospices performed well in management standards and there was no indication of urgent action needed. Work Engagement in our sample was higher compared with other National Health Service workers during the COVID-19 pandemic.

Psychological well-being of hospice staff: systematic review.

BACKGROUND: Poor psychological well-being among healthcare staff has implications for staff sickness and absence rates, and impacts on the quality, cost and safety of patient care. Although numerous studies have explored the well-being of hospice staff, study findings vary and the evidence has not yet been reviewed and synthesised. Using job demands-resources (JD-R) theory, this review aimed to investigate what factors are associated with the well-being of hospice staff. METHODS: We searched MEDLINE, CINAHL and PsycINFO for peer-reviewed quantitative, qualitative or mixed-methods studies focused on understanding what contributes to the well-being of hospice staff who provide care to patients (adults and children). The date of the last search was 11 March 2022. Studies were published from 2000 onwards in the English language and conducted in Organisation for Economic Co-operation and Development countries. Study quality was assessed using the Mixed Methods Appraisal Tool. Data synthesis was conducted using a result-based convergent design, which involved an iterative, thematic approach of collating data into distinct factors and mapping these to the JD-R theory. RESULTS: A total of 4016 unique records were screened by title and abstract, 115 full-text articles were retrieved and reviewed and 27 articles describing 23 studies were included in the review. The majority of the evidence came from studies of staff working with adult patients. Twenty-seven individual factors were identified in the included studies. There is a strong and moderate evidence that 21 of the 27 identified factors can influence hospice staff well-being. These 21 factors can be grouped into three categories: (1) those that are specific to the hospice environment and role, such as the complexity and diversity of the hospice role; (2) those that have been found to be associated with well-being in other similar settings, such as relationships with patients and their families; and (3) those that affect workers regardless of their role and work environment, that is, that are not unique to working in a healthcare role, such as workload and working relationships. There was strong evidence that neither staff demographic characteristics nor education level can influence well-being. DISCUSSION: The factors identified in this review highlight the importance of assessing both positive and negative domains of experience to determine coping interventions. Hospice organisations should aim to offer a wide range of interventions to ensure their staff have access to something that works for them. These should involve continuing or commencing initiatives to protect the factors that make hospices good environments in which to work, as well as recognising that hospice staff are also subject to many of the same factors that affect psychological well-being in all work environments. Only two studies included in the review were set in children's hospices, suggesting that more research is needed in these settings. PROSPERO REGISTRATION NUMBER: CRD42019136721 (Deviations from the protocol are noted in Table 8, Supplementary material).

Assessing the suitability of the Carer Support Needs Assessment Tool (CSNAT-Paediatric) for use with parents of children with a life-limiting condition: A qualitative secondary analysis.

BACKGROUND: The demands of caring for a child with a life-limiting condition can have a profound impact on parents' health and wellbeing. Currently, there is no standard procedure for identifying and addressing the support needs of these parents. AIM: To assess the suitability of the Carer Support Needs Assessment Tool (CSNAT (Paediatric)) for use with parents of children with a life-limiting condition. DESIGN: Secondary qualitative content analysis of two qualitative datasets exploring the health, wellbeing and experiences of support of mothers and fathers of children with a life-limiting condition. SETTING: A total of 30 mothers and 12 fathers were recruited via four UK children's hospices and social media. RESULTS: Parental experiences of support mapped onto the existing domains of the CSNAT (Paediatric). One aspect of their experience, surrounding their child's educational needs, went beyond the existing domains of the CSNAT. An adapted version of the tool CSNAT (Paediatric) should include this domain. CONCLUSION: The CSNAT (Paediatric) is a relevant tool for the assessment of parental support needs. Further research should assess the acceptability and feasibility of implementation of the broader intervention: CSNAT-I (Paediatric).

Safety, resource use and nutritional content of home-blended diets in children who are gastrostomy fed: findings from 'YourTube' - a prospective cohort study.

OBJECTIVE: To assess the risks, benefits and resource implications of using home-blended food in children with gastrostomy tubes compared with currently recommended formula feeds. DESIGN: This is a cohort study. Data were collected at months 0, 12 and 18 from parents and clinicians using standardised measures. SETTING: 32 sites across England: 28 National Health Service trusts and 4 children's hospices. PATIENTS: Children aged 6 months-18 years who were gastrostomy fed. MAIN OUTCOME MEASURE: The main outcome measure was the PedsQL Gastrointestinal Symptoms Scales score. Secondary outcomes included quality of life, sleep (child, parent), dietary intake, anthropometry, healthcare usage, safety outcomes and resource use. RESULTS: 180 children and families completed the baseline data collection, with 134 (74%) and 105 (58%) providing follow-up data at 12 and 18 months. There were fewer gastrointestinal (GI) symptoms at all time points in the home-blended diet group, but there was no difference in change over time within or between the groups. The nutritional intake of those on a home-blended diet had higher calories per kilogram and fibre, and both home-blended and formula-fed children have values above the dietary reference values for most micronutrients. Safety outcomes were similar between groups and over time. The total costs to the statutory sector were higher among children who were formula fed, but the costs of purchasing special equipment for home-blended food and the total time spent on childcare were higher for families with home-blended diet. CONCLUSIONS: Children who are gastrostomy fed a home-blended diet have similar safety profile, adequate nutritional intake and lower burden of GI symptoms than formula-fed children.Trial registration number ISRCTN13977361.

Work-related resources and demands predicting the psychological well-being of staff in children's hospices.

OBJECTIVES: This study assessed the work-related resources and demands experienced by children's hospice staff to help identify staff support systems and organizational practices that offer the most potential to prevent staff burnout and enhance well-being at work. METHODS: The relationships between individual and organizational characteristics, work-related resources and demands, and burnout and work engagement outcomes experienced by children's hospice staff were explored using two surveys: the Children's Hospice Staff survey, completed by UK children's hospice staff, and the Children's Hospice Organisation and Management survey, completed by the Heads of Care. We used structural equation modeling to assess the relationships between the variables derived from the survey measures and to test a model underpinned by the Job Demands-Resource (JD-R) theory. RESULTS: There were 583 staff responses from 32 hospices, and 414 participants provided valid data for burnout and work engagement outcome measures. Most participants were females (95.4%), aged 51-65 years old (31.3%), and had more than 15 years of experience in life-limiting conditions (29.7%). The average score for burnout was 32.5 (SD: 13.1), and the average score for work engagement was 7.5 (SD: 1.5). The structural model validity showed good fit. Demands significantly predicted burnout (b = 4.65, p ≤ 0.001), and resources predicted work engagement (b = 3.09, p ≤ 0.001). The interaction between resources and demands only predicted work engagement (b = -0.31, p = 0.115). Burnout did not predict work engagement (b = -0.09, p = 0.194). SIGNIFICANCE OF RESULTS: The results partly supported the JD-R model, with a clear association between resources and work engagement, even when the demands were considered. Demands were only directly associated with burnout. The findings also identified a set of the most relevant aspects related to resources and demands, which can be used to assess and improve staff psychological well-being in children's hospices in the UK.