Abstract
This study aims to identify the symptoms, concerns, and care priorities of children with life-limiting conditions and their families. A semi-structured qualitative interview study was conducted, seeking perspectives from multiple stakeholders on symptoms, other concerns, and care priorities of children and young people with life limiting and life-threatening conditions and their families. Participants were recruited from six hospitals and three children’s hospices in the UK. Verbatim transcripts were analysed using framework analysis. A total of 106 participants were recruited: 26 children (5–17 years), 40 parents (of children 0–17 years), 13 siblings (5–17 years), 15 health and social care professionals, 12 commissioners. Participants described many inter-related symptoms, concerns, and care priorities impacting on all aspects of life. Burdensome symptoms included pain and seizures. Participants spoke of the emotional and social impacts of living with life-limiting conditions, such as being able to see friends, and accessing education and psychological support. Spiritual/existential concerns included the meaning of illness and planning for an uncertain future. Data revealed an overarching theme of pursuing ‘normality’, described as children’s desire to undertake usual childhood activities. Parents need support with practical aspects of care to help realise this desire for normality.
Conclusion: Children with life-limiting conditions and their families experience a wide range of inter-related symptoms, concerns, and care priorities. A holistic, child-centred approach to care is needed, allowing focus on pursuit of normal childhood activities. Improvements in accessibility, co-ordination, and availability of health services are required to achieve this.
What is Known: • Existing evidence regarding symptoms, concerns, and care priorities for children with life-limiting conditions is largely limited to proxy-reported data and those with a cancer diagnosis. • Child-centred care provision must be directed by children’s perspectives on their priorities for care. | |
What is New: • Social and educational activities are more important to children with life-limiting conditions than their medical concerns. • A holistic approach to care is required that extends beyond addressing medical needs, in order to support children with life-limiting conditions to focus on pursuit of normal childhood activities. |
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Introduction
Worldwide there are approximately 21 million children and young people aged 0–19 years (hereafter ‘children’) with life-limiting and life-threatening conditions (hereafter ‘life-limiting’) [1]. Life-limiting conditions are those for which there is no hope of cure and from which children will die [2]. Life-threatening conditions are those for which curative treatment may be feasible but may fail [2].
Due to medical advances, increasing numbers of children are living with life-limiting conditions [3, 4]. However, provision of children’s palliative care varies geographically, and increased prevalence has not been met with an equivalent increase in healthcare resource [3, 5].
Palliative care for adults is effective and cost-effective, reducing unplanned admissions and futile treatments [6,7,8], while improving quality of life, care quality, and survival [9,10,11]. There are almost 400 conditions that affect children for which palliative care could be beneficial [3, 12]. However, evidence for effectiveness of children’s palliative care is limited in part due to a lack of a valid and reliable outcome measure [13, 14]. Development of such a measure has repeatedly been identified as a research priority [15,16,17]. A measure is in development in sub-Saharan Africa and Belgium, but primary data to inform measurement has not been generated outside Africa [18,19,20].
Outcome measure development for children with life-limiting conditions is complex due to differences in age and developmental stage, the range of conditions [12], and the role of family in care provision. To establish face and content validity, it is imperative to understand which symptoms and concerns matter the most. However, most studies focus on children with cancer [21], or rely on proxy reports of parent/carers (hereafter ‘parents’) or health and social care (healthcare) professionals [21]. This exclusion of children from participating in primary research directly contradicts the growing focus on children having agency, with a right to be involved in their own healthcare decisions [22, 23] as active partners in their healthcare, not passive recipients [22, 24]. This study aimed to identify the symptoms, concerns, and care priorities of children with life-limiting conditions and their families.
Methods
Study design
Semi-structured, qualitative interview study reported in accordance with the consolidated criteria for reporting qualitative studies (COREQ) [25].
Setting
Children, parents, and healthcare professionals were recruited from six hospitals and three children’s hospices within three UK countries.
Commissioners were recruited via recommendations from healthcare professionals and the UK’s national children’s palliative care advocacy charity.
Sampling and recruitment
Inclusion criteria
Children (5–17 years) with any life-limiting condition; parents/carers with a child < 18 years old with a life-limiting condition; siblings (5–17 years) of children with a life-limiting condition; healthcare professionals with > 6 months experience of caring for children with life-limiting conditions; commissioners of UK paediatric palliative care services.
Exclusion criteria
Children: unable to communicate via an in-depth interview, using ‘draw and talk’ or play methods or via their parents; speaks a language not supported by NHS translation services; currently enrolled in another study; unable to give consent/assent.
Parents/carers and siblings: unable to give consent/assent, speaks a language not supported by NHS translation services.
Purposive sampling was used to ensure maximum variation in key demographics such as age and condition. Given the heterogeneity of the sample, the concept of pragmatic saturation was used to determine the required sample size in order for the dataset to have the required diversity and depth to meet the aims and objectives of the study [26].
Data collection
Semi-structured interviews were conducted using a topic guide informed by a systematic review of symptoms and concerns in children with life-limiting conditions [21] and the World Health Organisation (WHO) definition of paediatric palliative care [27]. The topic guide was reviewed by the study steering group (healthcare professionals, parents, and researchers). Interviews were conducted by LC (experienced children’s palliative care nurse, new to qualitative research), AR (new to qualitative research), and DB (experienced qualitative researcher). All interviewers received training and supervision on conducting interviews with children, including communication, legal, and ethical issues.
Interviews commenced with demographic questions and children were asked about their hobbies and interests to build rapport. Play and drawing were used to aid interviews where required. The topic guide contained an open question asking participants to describe their/their child’s condition and how it affects their/their child’s life. Interviews with professionals asked about the main symptoms, concerns, and care priorities of children with life-limiting conditions. Probes ensured that all domains from the WHO definition of palliative care were discussed, while allowing participants to discuss what mattered most. Interviews were audio-recorded, transcribed verbatim, and pseudonymised.
Data analysis
Transcripts were analysed by LC, DB, AR, DH, and HS using deductive (from the WHO domains of palliative care [27]) and inductive coding [28, 29]. Analysis followed the five steps of framework analysis: familiarisation, constructing a thematic framework, indexing and sorting, charting and mapping/interpretation [28,29,30] using NVivo software (Version 12). Using framework analysis allowed the authors to compare and contrast the findings from each theme overall and by participant group. Regular meetings were held to discuss emerging themes and resolve any differences (20% of transcripts were independently coded by two researchers). RH, KB, and CES were consulted if discrepancies could not be resolved. Analysis was reviewed by the study steering group throughout the study.
Ethical approval
Ethical approval was granted by the Bloomsbury research ethics committee (HRA:19/LO/0033). Participants over 16 years old provided written informed consent. Those with parental responsibility provided written informed consent for participants < 16 years. Those < 16 years provided written assent.
Results
Participant characteristics
A total of 103 interviews were conducted (April 2019–September 2020) with 106 participants: 26 children, 40 parents, 13 siblings, 15 health and social care professionals and 12 commissioners (see Table 1). Two sets of parents and one set of siblings were interviewed together. ICD-10-chapter headings are reporting for pseudonymity as some children reported rare conditions. Most interviews were carried out face-to-face in a location of the participant’s choosing. Due to the COVID-19 pandemic, 13 interviews were conducted remotely (telephone or video call) [31].
Priority healthcare outcomes
The priority healthcare outcomes of children with life-limiting conditions and their families fitted into five themes—physical, spiritual and existential, emotional and psychological, social and practical, and pursuing normality. Table 2 shows these themes and the subthemes that comprise them. Illustrative quotes are presented in Tables 3, 4, and 5 and supplementary Table 1 (S1). Themes and subthemes were often closely inter-related.
Physical symptoms and concerns
All participants spoke of the importance of managing pain and other physical symptoms (such as seizures and infection), and the impact of multiple medical interventions. Symptom management and children being ‘comfortable’ was important to parents and professionals (T3Q1). Pain and other symptoms were often linked to other themes. For example, if physical symptoms were well managed, then children were more likely to be happy, have reduced anxiety, and be able to participate in normal childhood activities. Professionals discussed symptom management in relation to managing expectations of care and setting realistic goals (T3Q2). Seizures were particularly distressing and often described as difficult to manage by parents (T3Q3), sometimes being triggered by noise and over excitement (T3Q4), meaning siblings had to play quietly.
Participants from all groups spoke of the difficulties children had with eating and drinking. Some children described feeling under pressure to maintain weight (T3Q5), and others required artificial feeding. Healthy siblings spoke of feeling guilty about consuming treats in front of a sibling who was unable to eat (T3Q6).
Tiredness and fatigue were a concern for both children and parents. Parents spoke of lack of sleep and exhaustion which impacted on ability to care for their child (T3Q7). Children spoke of overwhelming fatigue causing lack of stamina and the need to take daytime naps (T3Q8).
Siblings and children with life-limiting conditions were very aware of changes in physical appearance which impacted on school attendance, seeing friends, and social activities (T3Q9).
Spiritual and existential
Professionals spoke of lack of confidence in discussing spiritual and existential issues (T3Q10). For some patients and families, faith offered a source of comfort (T3Q11, S1Q1), whereas for others, it was a potential cause of conflict (T3Q12). Some moved more towards faith, for example, by having their child christened ‘just in case’ (T3Q12). Faith was also important in decisions about future care, with one participant describing how hospital policy on death registration and care of the body conflicted with her own culture (T3Q14).
Participants from all groups spoke about the uncertainty surrounding length of life (T3Q15), with children wanting to plan for their future regardless of their prognosis (T3Q16). Children were often determined to overcome and survive (T3Q17, S1Q3). Parents spoke of adjusting their hopes and dreams for a child who would be unlikely to reach typical life-course milestones (T3Q18) and questioned the meaning of illness (‘why me/why my child?’) (T3Q19). They expressed a desire for their child to live life as fully as possible, to their full potential, experience relationships with others, and have things to hope for and look forward to (T3Q20).
Emotional and psychological
All participants described many psychological and emotional impacts of living with a life-limiting condition. Where children had been diagnosed during childhood, rather than at birth, they spoke of an awareness of being different and having different life experiences (T4Q21). For some siblings, their experience led to desires to pursue caring careers (T4Q22), while children with life-limiting conditions sought out others with similar experiences (T4Q23).
All participants spoke of the life-altering impact of living with a life-limiting condition (T4Q24). They described anger, worry, sadness (T4Q25, S1Q4), and an overwhelming desire for children to be happy (T4Q26, S1Q5). Older children spoke of loss of privacy, control, and independence (T4Q27–28, S1Q6–7). Parents also faced a loss of privacy due to having professionals in their home, and the wish to maintain some control over their child’s care and condition (T4Q29, S1Q8–9).
There was a sense of children and parents wanting to protect each other from how they were feeling, specifically around discussion of prognosis (T4Q30). Parents found accessing psychological support for themselves and siblings challenging, as this is often hospital-based and does not fit around work and school hours (T4Q31). Individuals also spoke of the importance of memory making (T4Q432).
Social concerns
Children were focused on being able to undertake usual childhood activities such as seeing friends, pursuing hobbies, and playing. School was important to parents and children for maintaining friendships, retaining a sense of normality and planning for a future by preparing for exams (T4Q33, S1Q10). Parents spoke of difficulty in accessing suitable education for their child due to complex medical needs (T4Q34). Many parents and children experienced loneliness and isolation due to absence from school and not being able to find suitable activities for their child to take part in (T4Q35–36, S1Q11). Unclear communication about symptom management goals and service availability often led to unrealistic expectations, causing discord between professionals and families. This impacted on decision-making, trust and respect, and continuity and co-ordination of care (T4Q37–38, S1Q12).
Parents and professionals spoke of the financial impact of having a child with a life-limiting condition in terms of having to give up work, the expense of hospital stays, and self-funding equipment due to lack of availability (T4Q39–40, S1Q13).
Practical concerns
Parents and professionals were concerned with many practical aspects of care. These included care quality, advance care planning, service availability and facilities (T5Q41–42, S1Q14), the huge familial burden of care, and the logistics of managing this (T5Q43, S1Q15). The physical burden of care increased as children grew older (T5Q44). Access to respite care was essential to many parents of children without a cancer diagnosis, allowing them to have uninterrupted sleep and spend time with other children (T5Q45). Children did not share these concerns and were more interested in being at home (not hospital), being able to see their friends and carry on with their usual activities (T5Q46–47, S1Q16–17).
Parents and children felt well informed about the condition, treatment, and available services, which was considered important (T5Q48, S1Q18). Siblings often felt less well informed and not included in care (T5Q49).
Normality
The theme of normality was cross-cutting across all other themes. Children wanted to live life as normally as possible, focusing on being a child first, with their condition secondary to this (T5Q50). They described the importance of seeing friends, attending school, and making plans for the future. To achieve this, physical symptoms need to be well managed. Children with varying diagnoses described normality in different ways, with all wanting to pursue normal childhood activities. When a condition had been present since birth or soon after, children spoke of feeling normal and not knowing any different (T5Q51). Those that had been diagnosed later in childhood spoke of having to adjust to a new normal such as having carers in the home (T5Q52). Those with an uncertain prognosis, such as cancer, wanted life to return to pre-diagnosis normality and desired to be like their healthy peers (T5Q53–54). Parents who had been caring for a child with a life-limiting condition for many years had often adjusted to their child’s care needs and had to remind themselves of their unique situation (T5Q55, S1Q19). Siblings spoke of seeing their unwell sibling as normal but with different needs (T5Q56, S1Q20).
Discussion
This study provides novel evidence of inter-related symptoms, concerns, and care priorities for children with a wide range of life-limiting conditions and their families, from the perspectives of multiple stakeholders (including children). This is an area of knowledge not previously well described [21]. Symptoms and concerns were broadly the same across the spectrum of life-limiting conditions, which is a finding previously reported [21, 32]. Most were evident across participant groups, except practical aspects of care, which were not a priority for children.
The concept of child-centred care encourages healthcare professionals to place the child and their interests at the centre of thinking and, where able, include them as active participants [22]. The focus of care is on the child in the context of the family, while acknowledging the child’s wider environment and relationships [22, 33]. Previous studies have found that children with cancer and their families try to adjust to a ‘new normal’, and those with severe neurological impairment were able to regain some normality with input from a paediatric palliative care team [34,35,36]. Our study adds to the concept of pursuing normality within the context of children living with life-limiting conditions, demonstrating that a child-centred approach to care needs to take an individual and holistic view of the child, ensuring that physical, emotional, social, practical, and spiritual concerns are addressed. This enables children to pursue normal childhood activities such as attending school and seeing friends. Children in our study wanted to be seen as children first, with their condition coming second to this, reinforcing that children do not want to be defined by their condition [37].
We found children wanted the opportunity to make plans for a longer-term future, even if these would not be realised, adding to the concept of pursuing normality. In contrast, a previous study found that children with neuro-disability only want to plan for the present or near future [38]. This difference may be due to the older age of the sample of participants with neurodisability meaning they had a better understanding of their condition. The heterogeneity of conditions in our study may also have contributed to our finding, as curative treatment for some life-limiting conditions is feasible, but may fail [39].
Taking a child-centred approach to care for children with life-limiting conditions needs to incorporate support for the family, while ensuring that the child remains the focus of care [40, 41]. This is important for families of children with life-limiting conditions, as this study demonstrates that they often have to provide complex, burdensome care. Many life-limited children are unable to communicate their needs due to their condition, and parents will need to advocate for their best interests. Parents require access to adequate holistic services, particularly respite care and practical support to enable them to provide care. Parents and siblings need time and space to undertake their own normal activities such as self-care, spending time as a family, and seeing friends. In our study, this was not always achieved, with insufficient or inaccessible practical, psychological, educational, and respite support often highlighted, along with lack of co-ordination and communication between services. To attempt to address this pursuit of normality and accomplish child-centred care, services need to be co-ordinated around child and family needs [40, 42], and this should be considered in the design of future health services for those with life-limiting conditions.
In our study, we found that children as young as five wanted to be informed about their condition, supporting a child-centred approach to care where the child is, where able, encouraged, and supported to be an active participant. Other studies have found that the desire to be informed about a condition is associated with adolescence, rather than younger children [21]. Siblings wanted to be informed, which is a finding previously reported in children whose parents have a life-limiting illness [43].
Strengths and limitations
As far as the authors are aware, this is one of the largest studies conducted exploring symptoms and concerns of children with a range of life-limiting conditions from multiple stakeholder perspectives. We have demonstrated that verbal children from the age of five years old are willing and able to participate in research and share their perspectives on their condition. This study’s strengths include our large sample, wide range of stakeholder participants, and the range of life-limiting conditions. Fathers, who are often underrepresented in palliative care research, represented 25% of our parent sample [44].
Our study has several limitations. Recruitment took place in a small number of UK sites and data on ethnicity was not collected. One site recruited only children with gastrointestinal diagnoses, and this is reflected in the higher number of participants from this group. There are almost 400 different life-limiting conditions known to affect children, so not all could be included [12]. Many children with life-limiting conditions are non-verbal and cannot meaningfully share their perspectives and parent/proxy-reporting has to be used. The findings presented here reflect those of children who were able to participate. As a child-centred approach to care should include support for the family, care must enable them to use their knowledge and experience of their child in order to advocate for them. The child’s needs and interests should always be at the centre of care and decisions [42].
Clinical and research implications
This study provides a comprehensive insight into what symptoms, concerns, and care priorities are important to children with life-limiting conditions and their families, to enable healthcare professionals to support them to be viewed as children, rather than their condition, within a child-centred model of care. We have demonstrated that children can be meaningfully involved in such studies [45]. Findings will be used to develop the construct for a valid child-centred outcome measure for use in this population.
Conclusions
Children want to focus on pursuing normal childhood activities, but need a holistic approach in addressing their care needs to achieve this. Improvements in accessibility, availability, and co-ordination of relevant health services are required.
Abbreviations
- Children:
-
Children and young people
- Healthcare professional:
-
Health and social care professional
- Life-limiting condition:
-
Life-limiting and life-threatening condition
- Parents:
-
Parents and carers
References
Connor SR, Downing J, Marston J (2017) Estimating the global need for palliative care for children: a cross-sectional analysis. J Pain Symptom Manage 53(2):171–177. https://doi.org/10.1016/j.jpainsymman.2016.08.020
Together for Short Lives (2019) Introduction to children’s palliative care. https://www.togetherforshortlives.org.uk/changing-lives/supporting-care-professionals/introduction-childrens-palliative-care/. Accessed 31 May 2022
Mitchell S, Morris A, Bennett K, Sajid L, Dale J (2017) Specialist paediatric palliative care services: what are the benefits? Arch Dis Child 102(10):923–929. https://doi.org/10.1136/archdischild-2016-312026
Fraser LK, Gibson-Smith D, Jarvis S, Norman P, Parslow RC (2020) Estimating the current and future prevalence of life-limiting conditions in children in England. Palliat Med. https://doi.org/10.1177/0269216320975308
Together for Short Lives (2019) A workforce in crisis: children’s palliative care in 2019. Bristol: Together for Short Lives
Gomes B, Calanzani N, Curiale V, McCrone P, Higginson IJ (2013) Effectiveness and cost-effectiveness of home palliative care services for adults with advanced illness and their caregivers. Cochrane Database Syst Rev (6):CD007760
Gomes B, Calanzani N, Higginson IJ (2014) Benefits and costs of home palliative care compared with usual care for patients with advanced illness and their family caregivers. JAMA 311(10):1060–1061. https://doi.org/10.1001/jama.2014.553
Higginson IJ, Evans CJ (2010) What is the evidence that palliative care teams improve outcomes for cancer patients and their families? Cancer J 16(5):423–435. https://doi.org/10.1097/PPO.0b013e3181f684e5
Temel JS, Greer JA, Muzikansky A, Gallagher ER, Admane S, Jackson VA et al (2010) Early palliative care for patients with metastatic non-small-cell lung cancer. N Engl J Med 363(8):733–742. https://doi.org/10.1056/NEJMoa1000678
Smith S, Brick A, O’Hara S, Normand C (2014) Evidence on the cost and cost-effectiveness of palliative care: a literature review. Palliat Med 28(2):130–150. https://doi.org/10.1177/0269216313493466
Farquhar MC, Prevost AT, McCrone P, Brafman-Price B, Bentley A, Higginson IJ et al (2014) Is a specialist breathlessness service more effective and cost-effective for patients with advanced cancer and their carers than standard care? Findings of a mixed-method randomised controlled trial. BMC Med 12:194. https://doi.org/10.1186/s12916-014-0194-2
Hain R, Devins M, Hastings R, Noyes J (2013) Paediatric palliative care: development and pilot study of a ‘Directory’ of life-limiting conditions. BMC Palliat Care 12(1):43. https://doi.org/10.1186/1472-684x-12-43
Dawson J, Doll H, Fitzpatrick R, Jenkinson C, Carr AJ (2010) The routine use of patient reported outcome measures in healthcare settings. BMJ 340:c186. https://doi.org/10.1136/bmj.c186
Coombes LH, Wiseman T, Lucas G, Sangha A, Murtagh FE (2016) Health-related quality-of-life outcome measures in paediatric palliative care: a systematic review of psychometric properties and feasibility of use. Palliat Med 30(10):935–949. https://doi.org/10.1177/0269216316649155
Friedel M, Aujoulat I, Dubois A-C, Degryse J-M (2018) Instruments to measure outcomes in pediatric palliative care: a systematic review. Pediatrics e20182379. https://doi.org/10.1542/peds.2018-2379
Medical Research Council (2009) Patient- reported outcome measures (PROMs): identifying UK research priorities. MRC, London
Booth A, Maddison J, Wright K, Fraser L, Beresford B (2018) Research prioritisation exercises related to the care of children and young people with life-limiting conditions, their parents and all those who care for them: a systematic scoping review. Palliat Med 32(10):1552–1566. https://doi.org/10.1177/0269216318800172
Downing J, Atieno M, Powell RA, Ali Z, Marston J, Meiring M et al (2012) Development of a palliative care outcome measure for children in sub-Saharan Africa: findings from early phase instrument development. Eur J Palliative Care 19(6):4
Downing J, Namisango E, Harding R (2018) Outcome measurement in paediatric palliative care: lessons from the past and future developments. Ann Palliat Med 7(Suppl 3):S151–S163. https://doi.org/10.21037/apm.2018.04.02
Friedel M, Brichard B, Boonen S, Tonon C, De Terwangne B, Bellis D et al (2020) Face and content validity, acceptability, and feasibility of the adapted version of the children’s palliative outcome scale: a qualitative pilot study. J Palliat Med. https://doi.org/10.1089/jpm.2019.0646
Namisango E, Bristowe K, Allsop MJ, Murtagh FEM, Abas M, Higginson IJ et al (2019) Symptoms and concerns among children and young people with life-limiting and life-threatening conditions: a systematic review highlighting meaningful health outcomes. Patient 12(1):15–55. https://doi.org/10.1007/s40271-018-0333-5
Ford K, Dickinson A, Water T, Campbell S, Bray L, Carter B (2018) Child centred care: challenging assumptions and repositioning children and young people. J Pediatr Nurs 43:e39–e43. https://doi.org/10.1016/j.pedn.2018.08.012
UN General Assembly (1989) Convention on the Rights of the Child. UN
Wijngaarde RO, Hein I, Daams J, Van Goudoever JB, Ubbink DT (2021) Chronically ill children’s participation and health outcomes in shared decision-making: a scoping review. Eur J Pediatr 180(8):2345–2357. https://doi.org/10.1007/s00431-021-04055-6
Tong A, Sainsbury P, Craig J (2007) Consolidated criteria for reporting qualitative research (COREQ): a 32-item checklist for interviews and focus groups. Int J Qual Health Care 19(6):349–357. https://doi.org/10.1093/intqhc/mzm042
Low J (2019) A pragmatic definition of the concept of theoretical saturation. Sociol Focus 52:1–9. https://doi.org/10.1080/00380237.2018.1544514
World Health Organisation (2013) WHO Definition of Palliative Care. https://www.who.int/cancer/palliative/definition/en/ Accessed 31 May 2022
Gale NK, Heath G, Cameron E, Rashid S, Redwood S (2013) Using the framework method for the analysis of qualitative data in multi-disciplinary health research. BMC Med Res Methodol 13:117. https://doi.org/10.1186/1471-2288-13-117
Ritchie J, Lewis J, McNaughton-Nicholls C, O’rmston RT (2014) Qualitative research practice: a guide for social science students and researchers. London: Sage
Miles MB, Huberman AM (2002) The qualitative researcher’s companion: classic and contemporary readings. SAGE, London
Scott HM, Coombes L, Braybrook D, Roach A, Harðardóttir D, Bristowe K et al (2022) COVID-19: impact on pediatric palliative care. J Pain Symptom Manage. https://doi.org/10.1016/j.jpainsymman.2022.02.330
Feudtner C, Nye R, Hill DL, Hall M, Hinds P, Johnston EE et al (2021) Polysymptomatology in pediatric patients receiving palliative care based on parent-reported data. JAMA Netw Open 4(8). https://doi.org/10.1001/jamanetworkopen.2021.19730
Southall DP, Burr S, Smith RD, Bull DN, Radford A, Williams A et al (2000) The Child-Friendly Healthcare Initiative (CFHI): healthcare provision in accordance with the UN Convention on the Rights of the Child. Child Advocacy International. Department of Child and Adolescent Health and Development of the World Health Organization (WHO). Royal College of Nursing (UK). Royal College of Paediatrics and Child Health (UK). United Nations Children’s Fund (UNICEF). Pediatrics 106(5):1054–64. https://doi.org/10.1542/peds.106.5.1054
Ribbers S, Wager J, Hartenstein-Pinter A, Zernikow B, Reuther M (2020) Core outcome domains of pediatric palliative care for children with severe neurological impairment and their families: a qualitative interview study. Palliat Med 34(3):309–318. https://doi.org/10.1177/0269216319885818
Beecham E, Langner R, Hargrave D, Bluebond-Langner M (2019) Children’s and parents’ conceptualization of quality of life in children with brain tumors: a meta-ethnographic exploration. Qual Health Res 29(1):55–68. https://doi.org/10.1177/1049732318786484
Earle EA, Clarke SA, Eiser C, Sheppard L (2007) ‘Building a new normality’: mothers’ experiences of caring for a child with acute lymphoblastic leukaemia. Child Care Health Dev 33(2):155–160. https://doi.org/10.1111/j.1365-2214.2006.00638.x
Mitchell S, Slowther A-M, Coad J, Dale J (2020) Experiences of healthcare, including palliative care, of children with life-limiting and life-threatening conditions and their families: a longitudinal qualitative investigation. Arch Dis Childhood. archdischild-20. https://doi.org/10.1136/archdischild-2020-320189
Allard A, Fellowes A, Shilling V, Janssens A, Beresford B, Morris C (2014) Key health outcomes for children and young people with neurodisability: qualitative research with young people and parents. BMJ Open 4(4):e004611. https://doi.org/10.1136/bmjopen-2013-004611
Together for Short Lives (2018) A guide to children’s palliative care. Bristol: Together for Short Lives
Department of Health (2003) Getting the right start: national service framework for children ; standard for hospital services. National service framework assessments. London: Department of Health
Coyne I, Hallström I, Söderbäck M (2016) Reframing the focus from a family-centred to a child-centred care approach for children’s healthcare. J Child Health Care 20(4):494–502. https://doi.org/10.1177/1367493516642744
Carter B, Bray L, Dickinson A, Edwards M, Ford K (2014) Child-centred nursing: promoting critical thinking. London: SAGE
Marshall S, Fearnley R, Bristowe K, Harding R (2021) The perspectives of children and young people affected by parental life-limiting illness: an integrative review and thematic synthesis. Palliat Med 35(2):246–260. https://doi.org/10.1177/0269216320967590
Nicholas D, Beaune L, Belletrutti M, Blumberg J, Ing S, Rapoport A et al (2020) Engaging fathers in pediatric palliative care research. J Soc Work End Life Palliat Care 16(1):42–56. https://doi.org/10.1080/15524256.2019.1703877
Roach A, Braybrook D, Marshall S (2021) Reflective insights from developing a palliative care children and young people’s advisory group. Palliat Med 35(3):621–624. https://doi.org/10.1177/0269216320976035
Acknowledgements
The Children’s Palliative care Outcome Scale (CPOS) Study Steering Group members are as follows: AK Anderson, Jo Bayly, Lydia Bates (PPI), Debbie Box, Rachel Burman, Lizzie Chambers, Alan Craft, Finella Craig, Aislinn Delaney, Jonathan Downie, Sara Fovargue, Jane Green (PPI), Jay Halbert, Julie Hall-Carmichael, Irene Higginson, Michelle Hills, Mevhibe Hocaoglu, Vanessa Holme, Gill Hughes, Joanna Laddie, Angela Logun (PPI), Eve Malam, Steve Marshall, Linda Maynard, Andrina McCormack, Catriona McKeating, Lis Meates, Eve Namisango, Veronica Neefjes, Cheryl Norman, Susan Picton, Christina Ramsenthaler, Ellen Smith, Michelle Ward, Frances Waite, Mark Whiting. This study is supported by the National Institute for Health Research (NIHR) Applied Research Collaboration South London (NIHR ARC South London) at King’s College Hospital NHS Foundation Trust.
Funding
CPOS was funded by a European Research Council’s Consolidator Award (grant ID: 772635) with the overall aim to develop and validate a person-centred outcome measure for children, young people, and their families affected by life-limiting and life-threatening condition.
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All authors: conception and design of the work. LC, DB and AR: data collection. LC, DB, AR, DH, HS: data analysis. All authors: interpretation of data. LC draft of paper. All authors: critical review and revision of article.
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Ethical approval was granted by the Bloomsbury research ethics committee (HRA:19/LO/0033).
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Participants over 16 years old provided written informed consent. Those with parental responsibility provided written informed consent for participants < 16 years. Those < 16 years provided written assent.
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Principal Investigator: Richard Harding. This article reflects only the authors’ views and the European Research Council is not liable for any use that may be made of the information contained therein. Fliss Murtagh is a National Institute for Health Research (NIHR) Senior Investigator. The views expressed in this article are those of the author and not necessarily those of the NIHR, or the Department of Health and Social Care.
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Coombes, L., Braybrook, D., Roach, A. et al. Achieving child-centred care for children and young people with life-limiting and life-threatening conditions—a qualitative interview study. Eur J Pediatr 181, 3739–3752 (2022). https://doi.org/10.1007/s00431-022-04566-w
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DOI: https://doi.org/10.1007/s00431-022-04566-w