Decision-making regarding place of end-of-life care for children with life-limiting and life-threatening conditions: a systematic integrative review

Background

There has been a marked increase in the number of children living with life-limiting and life-threatening conditions, e.g., cancer, genetic disorders, and neurological conditions [1, 2]. In England, for example, the prevalence of children with a life-limiting condition rose from 26.7 per 10,000 in 2001/02 to 66.4 per 10,000 in 2017/18 [2]. This is largely due to improvements in technology, treatments, and medical expertise [3]. Future predictions estimate that this trend will continue [2]. Thus, more children and their families will require palliative and end-of-life care in the future. Globally, there is growing recognition of the need to optimise information on available palliative care services and to enhance communication between acute and community-based services for children with life-limiting and life-threatening conditions and their families [4–6].

A child’s death has a long-lasting and traumatic impact on families and healthcare professionals [7, 8]. Consequently, when death in childhood is anticipated, such as when a child is living with a life-limiting or life-threatening condition, it is important to provide high-quality care to maximise quality of life and facilitate end-of-life care and death in the preferred place [9]. ‘Home’ is often the preferred place for many families when their child needs palliative and end-of-life care, though this preference can change due to factors such as the child’s condition, availability of palliative care services, and family circumstances [10]. However, the evidence around best practices for decision-making about the place of end-of-life care remains inadequate [11]; some families may prefer alternatives to home [11]. Additionally, most children with life-limiting and life-threatening conditions continue to die in hospital settings [11]. Papadatou et al. recommend that policymakers work to improve end-of-life care in all settings since home, hospital, and hospice, as places of care, are desirable to different families for different reasons at different times [12]. As a result, the place of end-of-life care has been identified as a research priority to guide healthcare services and inform service development [9, 13].

Several quantitative retrospective cohort studies to date have investigated end-of-life care for children with life-limiting and life-threatening conditions [11, 14–16]. While these studies have yielded important information about place of end-of-life care by retrieving data from medical records, gaps in knowledge remain in relation to understanding factors influencing decisions around why a particular place (i.e., hospital, hospice, home) is chosen for end-of-life care [11, 14–16]. Other studies have focused on place of end-of-life care solely for children with advanced cancer [17–20]. The emphasis so far on advanced cancer is reflected in the qualitative systematic review by Noyes et al. which synthesised factors that influence parental decision-making when choosing place of end-of-life care and death for children with cancer [21]. This review provides valuable insights into decision-making around location of end-of-life care for children with cancer, however, it remains unknown whether the findings are transferable to a range of childhood life-limiting and life-threatening conditions. Another recent scoping review investigated the importance of place in paediatric palliative care for families (from the parent’s perspective) [22]. This scoping review identified a broad range of factors that contribute to the choice of place for end-of-life care, including familial support, parent capabilities, access to resources, and proximity to medical care, but did not include health and social care professionals (HSCPs) perspectives [22]. However, to our knowledge, no systematic integrative review has been undertaken to examine decision-making about place of end-of-life care for children with a wider range of conditions from the perspectives of parents, children, and HSCPs. Therefore, this integrative review aimed to synthesise the evidence base on factors influencing decision-making on place of end-of-life care for children with life-limiting and life-threatening conditions.

Methods

The methodological guidelines and procedures for conducting systematic integrative reviews as outlined by Whittemore and Knafl [23] were followed. The review was conducted and reported in accordance with the Preferred Reporting Items for Systematic Review and Meta-Analysis (PRISMA) 2020 guidelines [24] (see Additional file 1 for PRISMA checklist). The protocol for this review was registered with PROSPERO (No. CRD42023406800) [25].

Results

As illustrated in Fig. 1, database searches yielded 451 studies after duplicates were removed. Of these, 391 did not meet the eligibility criteria following title and abstract screening. A total of 60 studies met the criteria for full text review, and at this stage, a further 49 studies were excluded, with reasons noted in Fig. 1. In total, 11 studies were deemed eligible for inclusion in this review.

Discussion

This integrative review systematically identified and synthesised the available evidence on factors influencing decision-making regarding place of end-of-life care for children (≤ 18 years) with life-limiting and life-threatening conditions. This review provides nuanced insights into multiple complex factors that influence decision-making on choosing place of end-of-life for a child.

Previous research indicated that home is the preferred place for end-of-life care for both children and parents [9, 34]. Our review findings suggest that the child’s condition, including symptom severity, time to death, and risk of deterioration, is an influential factor considered by parents and HSCPs when choosing home as the place for the child’s end-of-life care. Previous literature has revealed that parents desire the child’s pain and symptoms to be controlled, but parents may lack the confidence to manage this themselves at home [38–40]. The data in our review indicated that the child’s preference was to be at home [12, 29, 35]. This created a complex balance for parents to strike between honouring the child’s wishes whilst also considering the practicalities of pain and symptom control.

Our review also revealed that where there were limited home or community care services for end-of-life care, parental capacity and skill set to care for their child impacted whether the child could go, or remain at, home for end-of-life care. Previous research shows that limited access to services in some geographical areas is a significant issue that impacts on decision-making for place of end-of-life care [41]. This is further supported by an overview of palliative care in Europe which highlights inequality between countries and regions regarding policy and service provision [42]. This variation suggests varying opportunities for families to make meaningful decisions concerning place of palliative care for their child and place for a peaceful death. Our review highlights the need for system-wide improvements and policy strategies that empower parents to choose the most suitable end-of-life care setting for their child and family regardless of parental skill set.

Our review revealed situations where parents were given no choice about place of end-of-life care for their child or where a lack of information provided made parents feel as if there was no choice. In such situations, parents’ control was obliterated, and their capacity was significantly diminished. Previous research has indicated that information provision to parents has clear value in helping them make informed decisions [43–47].

The wellbeing of siblings was considered alongside the wellbeing of the child receiving end-of-life care when choices were being made about place for end-of-life care. Key findings in this review were safeguarding siblings, facilitating sibling connectedness, maintaining family life, and familiarity and comfort of home. Previous research found that parents of children with life-limiting conditions often felt they prioritised their unwell child’s needs, resulting in siblings sometimes feeling deprioritised [48]. Other research however has shown that siblings of unwell children receiving palliative care when directly spoken to consider themselves part of a special and happy family [49].

The findings of our review highlight a lack of research studies specifically investigating the impact of choice of place of end-of-life care on the wellbeing of siblings of children receiving palliative and end-of-life care. Furthermore, our review highlighted that there can be contrasting attitudes from parents in how to best meet the needs of siblings, and that this is an influential factor in parent’s decision-making. For instance, some parents desire to protect their children from death, or possibly from the suffering that their dying child is going through [12, 29]. Notwithstanding this, siblings often demonstrate an awareness that death is coming, and desire to be involved [49–51], illustrating that sibling inclusion in decision-making about place of end-of-life care requires further investigation. Future research to understand sibling perspectives to inform the development of bespoke family-focused interventions to support advanced care planning for place of end-of-life care for children with life-limiting and life-threatening conditions is required.

Our review highlighted that there are limited studies examining the child’s experience of, and involvement in, deciding place of their end-of-life care. One existing research article largely focused on understanding children with cancer’s decision-making about their care and treatment while undergoing palliative care [52], and one other article included a discussion to inform policy for prospective decision-making [53]. Strategies for communication with children about cancer diagnosis and treatment have for instance been highlighted in the literature, but these strategies are poorly implemented [54, 55]. This lack of children’s voices regardless of diagnosis in deciding their place of end-of-life care supports previous research which has highlighted a gap in hearing children’s voices in relation to healthcare provision, including end-of-life care [56]. It has been documented that including children’s voices may present challenges given the nature of some conditions (e.g., curative possibility of cancer) [54], the sensitivity of discussing end-of-life care and parental desire to protect children from the possibility of death. Notwithstanding this future research might be of benefit to understand how to meaningful and ethically involve children who are receiving end-of-life care in palliative care research.

Our review also highlighted the lack of evidence of HSCP experiences of decision-making about place of end-of-life care for children. Shared decision-making is recommended in international guidance documents written for HSCPs [44, 57]. HSCPs bring to shared decision-making an in-depth understanding of family values, and this helps them provide end-of-life care aligned to family need [57]. Positive relationships between HSCPs and parents have been shown to support parents making end-of-life decisions, both in our review and elsewhere [58]. Thus, HSCPs developing positive relationships with parents/families, would assist families in making decisions about the place of end-of-life care for their child. Further to this, what is unknown from the current synthesis is an understanding of HSCPs decision-making process when considering where end-of-life care should take place. Ultimately there is the need for more empirical research studies to further investigate and understand the perspectives of children, parents, and HSCPs when deciding upon the place of end-of-life care for the child [59].

Conclusions

This review found that there are a complex range of factors to be considered in practice, largely by parents, when deciding on place of end-of-life care for children with life-limiting and life-threatening conditions. The evidence from this review demonstrates the need for guidance and policies to enable system-wide improvements and to empower parents to choose to be actively involved in deciding what place of end-of-life care is best for their child and family. This review found that some parents are not afforded a choice when it comes to decision-making about place of end-of-life care. It is important for parents to be involved in decisions about place of end-of-life care for their child and for parents to be provided with information to help them in the decision-making process. When this is not the case, families may feel unsupported, and the care provided may not reflect their preferences. There are clear benefits to advance care planning, as sensitive, timely, and honest discussions can help facilitate the wishes of families and ensure better support during this challenging time. It is evident from this review that parents consider the impact on the whole family, including siblings, when making decisions. Thus, HSCPs should be mindful of the child’s family when influencing decisions about place of end-of-life care. Finally, future research needs to evaluate HSCP experience of decision-making about place of end-of-life care, but also, crucially, consider how to meaningfully involve the child themselves.

Supplementary Information

Authors’ contributions

Conceptualization: V.L., P.M., J.R., J.P., Y.C., G.K., T.M., H.K., E.C.; F.H., AC Formal analysis: F.H., A.C. with support from G.K., H.K.; Investigation: A.C., F.H., G.K.; Methodology: All authors; Data curation: A.C., F.H.; Writing—original draft: A.C., F.H.; Writing – review & editing: All authors, with critical review and final edits by V.L.; Supervision: G.K., H.K., P.M., V.L.; Project administration: V.L., P.M., A.C., F.H.; Funding acquisition: V.L., P.M., J.R., J.P., Y.C., G.K., T.M., H.K., E.C..

Funding

This programme of research is supported by the Higher Education Authority (HEA) North–South Research Programme, Shared Island Fund, on behalf of the Department of Further and Higher Education, Research Innovation and Science (DFHERIS) [Grant ID: CO-DECIDE].

Data availability

No datasets were generated or analysed during the current study.

Declarations