A key aim of the study was to examine expert opinion on the integration and coordination of current services including palliative care for children with LLNDD and their families in Ireland. The findings suggest that services do not serve this group well, with deficiencies in care compounded by a lack of information on available services and sub-optimal communication between settings. These findings reflect previous research with professionals in other pediatric settings8,28. The achievement of quality of life (rather than prolongation of life) was strongly articulated as the primary goal of care for children with LLNDD in keeping with an overall palliative approach to the care of these children. However, it was the opinion of the panel that palliative care is considered relatively late in the child’s condition or in crisis management. This is at odds with the current trend towards early integration of palliative for adults and children seen internationally. Despite the clarity of these statements, the lack of consensus when considering other aspects of palliative care was notable, pointing to different experiences of and opinions regarding provision in Ireland. Diversity of opinion was evident when considering such matters regarding communication with parents, recognition of palliative care needs and adequacy of access to specialist palliative care.
Perhaps unsurprisingly, the panel’s recommendations regarding priority changes that would improve care are closely tied to statements that reached consensus in the early sections of the Delphi questionnaire. The top three priority changes to improve current services (i.e., a single care plan, better communication, assignment of key workers) are consistent with the expert panel’s agreement that there is inadequate information, poor communication and suboptimal access to services. Previous research has suggested that key-worker availability would improve coordination and integration of services to the child and family28;29;30 and it appears that the expert panel were of a similar opinion.
Even though quality of life was felt to be the primary goal of care for children with LLNDD and that inadequacies in palliative care provision were noted, strategies to improve palliative care provision did not gain consensus. The ranking that the strategies were given in the service improvement priority list indicated that some respondents did regard them as having some importance. Nevertheless, this was not the opinion of a sufficient majority. Our research did not explore reasons why those recommendations did not reach consensus, but barriers to the integration of palliative care in children’s care are well described in the literature31;32;33. It is possible that members of the expert panel share some of those viewpoints regarding the place of palliative care in children’s care, or it may be that they did not consider the strategies presented in the Delphi were the right ones to advance service provision. Delphi studies can help uncover social dimensions to decision-making and the lack of agreement on palliative care highlights an area that merits further exploration.
The centrality of a coordinated and multiagency approach to the planning and delivery of care and support to all children with palliative care needs has been documented34;35;1. Campbell36 identifies that in the context of disability services the highest quality ratings are achieved when there is evidence of the use of multidisciplinary integrated care pathways which clarify expected steps and outcomes. Similarly, the priority awarded to a keyworker for every family is consistent with the panel’s agreement that the current lack of this resource results in the ad hoc delivery of services. This is consistent with findings of previous research which suggest that key-worker availability would improve coordination and integration of services to the child and family28;29;30.
A key issue here appears to be the inconsistency between the panel’s opinion on service coordination and the proposal of “improved service coordination and integration” as a change that would improve services and the opinion of health professionals in Quinn et al’s28 study of professionals’ opinions about service integration and coordination for a generic population of life-limited children in the context of specialist palliative care. Another inconsistency relates to the panel’s proposal for the appointment of a specialist palliative care consultant as a priority change to services that would improve care. This appears inconsistent with the panel’s opinion on the current provision of palliative care to children with LLNDD, especially the panel’s failure to agree on issues of access to specialist palliative care. One explanation for this may be the difference between having access to an adult specialist palliative care service adapted to the needs of a child with a LLNND as is currently available, and access to a specialist pediatric palliative care service, which would be focused exclusively on the needs of the child and family, as is the proposed change to current services. In addition to specialist services, there was no consensus (despite high levels of agreement) on the potential contribution of GPs to care for this group. In the context of a lack of agreement of both generalist and specialist support for these services, there may be a vacuum about how to structure services.
While it has been clearly established that children’s needs from palliative care differs from that of adults (World Health Organisation, 2018), there are benefits to examining how the issues raised above have been considered in the adult palliative care literature. Looking to this literature, it seems that the issues of late identification and a lack of integrated services are also present here (Gomez-Batiste et al, 2017). However, several initiatives are evident internationally that have sought to addresses these issues in adult settings. Gomez-Batiste et al, 2017 report a number of European initiatives that have worked to address these issues in adult settings, including the issue of tools to promote early identification and prevalence of need and proposals for improving the delivery of palliative care approach in health service settings. Despite these initiatives, it is clear that there remain significant outstanding issues in adult care. Nevertheless, developments along these lines in children’s services would no doubt begin to address the issues identified in the present study. Indeed, the need to integrate children’s palliative into pediatric care is noted by the WHO (2018).
Strengths and Limitations
The findings of this study should be considered in the context of its strengths and limitations. There is little definitive guidance to be found in the literature with regards to the decisions made during a Delphi study, and although the decisions made, and the rationale for them, have been made clear in this study, it reflects the positive and negative contributions of the Delphi method.
The study used a relatively small expert panel when compared to the panel size in much published literature, however this may reflect the small expert pool available in Ireland. In this study, every effort was made to ensure this was a comprehensive panel, which included all services involved in the care of children with LLNDD and their families, nonetheless the findings should be considered in the context of the panel size. Nevertheless, the discrepancies between the perception of service coordination and proposed changes to services may be associated with the small number of experts. It is also possible that poor coordination of services in not a national problem and that service integration and coordination is worse in some areas than in others
It is also difficult to directly compare the response rate with other published literature due to ambiguity in what is being reported. Gibson, Koepsell, Diehr and Hale39 reported a 64% response rate, while Butterworth and Bishop40 report a response rate of 61%, however it is not clear whether this figure represents the respondents who completed all rounds. Sumsion27 suggests a minimum of 70% for each round. However, commonly this minimum response rate is not achieved, with a range of 40 – 65% reported in the literature14;41;42;15. This study met Sumsion’s standard for all but the first round.
It is the explicit and stated aim of Irish health policy to provide health services to all who need them. Despite these stated visions and aspirations, developing systems of services to best meet the needs of young children with LLNDD and their families continues to represent a significant challenge for all services and agencies involved in the care of this population. As noted in the Introduction, data collection for this study preceded several developments in policy with implications for supports to children with LLNDDs and their families. The PDSCYP program10 and the recommendations of the National Model of Care for Paediatric Services in Ireland11 aims to maximize access to specialist care close to home for children with complex needs. However, the need to fully implement these policies has been noted by researchers in the area43. So, while these documents provide a framework within which many of the issues noted in the present study can be addressed, the process of operationalizing change is ongoing. While not explored in this study, further research exploring healthcare professionals views regarding service improvements would further contribute to this process.
The present study’s findings highlight several possible recommendations for the care and support of children with LLNDD and their families, with the goal of achieving the best possible quality of life for the child and achievement of their full potential within the limits of the illness. With the focus on coordinated and integrated services noted in the findings, the practical operation of assigning a key worker to every family and developing a single care plan for the family, would contribute to achieving this. This would be further complemented by a focus on open and honest communication with the family and between health care professionals. Indeed, both elements are part of the Progressing Disabilities program and reflect the emphasis of this program on family centered care44. Reflecting on the other priorities of care reported in this study, the findings highlight the potential contribution of palliative care for this target group. The need for optimum symptom management, allowing the child to be cared for at home, would be supported by increased accessed to specialist pediatric palliative care as a resource when required. There is no doubt that pediatric palliative care services in Ireland are not as developed as those available for adults, but developments in service provision such as those described in this article may ensure additional support. However, even in countries where pediatric palliative care is more developed45, there is no guarantee that these services would be part of an integrated service network accessible to children with LLDNN and their families.