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Abstract
Background The number of children and young people (CYP) living with life-limiting and life-threatening conditions is rising. Paediatric palliative care is a relatively new aspect of healthcare, the delivery of which is variable, with a wide range of healthcare and voluntary sector providers involved. Policy recommendations are for Specialist Paediatric Palliative Care (SPPC) services to be supported by a physician with specialist training.
Aim To examine the research evidence regarding the distinct benefits of SPPC services, with ‘Specialist Paediatric Palliative Care’ defined as palliative care services supported by a specialist physician.
Method Systematic review of studies of SPPC services published in English from 1980 to 2016. Keyword searches were carried out in medical databases (Cochrane, PubMed, EMBASE, CINAHL and AMED) and a narrative synthesis.
Results Eight studies were identified, most of which were retrospective surveys undertaken within single institutions; three were surveys of bereaved parents and three were medical notes reviews. Together they represented a heterogeneous body of low-level evidence. Cross-cutting themes suggest that SPPC services improve the quality of life and symptom control and can impact positively on place of care and family support.
Conclusions Current evidence indicates that SPPC services contribute beneficially to the care and experience of CYP and their families, but is limited in terms of quantity, methodological rigour and generalisability. Further research is necessary given the significant workforce and resource implications associated with policy recommendations about the future provision of SPPC and to address the need for evidence to inform the design and delivery of SPPC services.
- Palliative Care
- Paediatric Practice
- Paediatric Staffing
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What is already known on this topic?
The number of children and young people (CYP) living with life-limiting and life-threatening conditions is rising with continuing advances in clinical medicine.
There are international recommendations and standards for Specialist Paediatric Palliative Care (SPPC) services, but this is a relatively new subspecialty and is inconsistently available.
The more universal adoption of recommendations and standards requires significant investment of resource, which is difficult to achieve.
What this study adds?
This is the first systematic review of research related to the evaluation of SPPC.
The review identifies a summary of the evidence that suggests that SPPC provides benefit to CYP and families.
Key themes have been identified to inform future service development and research in paediatric palliative care.
Introduction
With advances in medical treatments and the use of medical technology, a growing number of children and young people (CYP) live with life-limiting conditions (LLCs) and life-threatening conditions (LTCs).1 ,2 This includes CYP who live with conditions where curative treatment is feasible but can fail, while for others, there is no known cure.3 Ensuring sustainable healthcare services that can effectively meet the needs of these CYP and those of their families presents a significant challenge.
Palliative care for CYP has been defined as “an active and total approach to care, from the point of diagnosis or recognition, embracing physical, emotional, social and spiritual elements through to death and beyond. It focuses on enhancement of the quality of life for the CYP and support for the family and includes the management of distressing symptoms, provision of short breaks and care through death and bereavement.3” Globally, it is estimated that seven million CYP (aged 0–19) could benefit from palliative care services,4 with at least 49 000 in the UK.5 However, most countries have no paediatric palliative care services, including hospices.4 Where paediatric palliative care services have developed, this has been largely as a result of the determination of motivated individuals and charitable funders.6 ,7 The type and availability of services vary geographically due to the wide range of healthcare and voluntary sector providers involved.4
Specialist Paediatric Palliative Care (SPPC) services are defined in UK and European standards as those supported by a physician with specialty training (a consultant) in paediatric palliative medicine.1 ,8–10 However, SPPC is yet to become an established medical subspecialty, and few countries have doctors trained to this level.4 As a result, there is a tension between this standard of care and the many existing services which specialise in the provision of paediatric palliative care but lack the support of specialty-trained physicians.
Objective
The objective of this systematic review is to contribute to the debate regarding the design of paediatric palliative care services by specifically examining SPPC, defined as a palliative care service supported by a physician with specialty training in paediatric palliative medicine, and asking “What are the distinct benefits of these SPPC services to CYP and their families?” The review also provides an opportunity to identify evidence gaps for further research.
Design
The Centre for Reviews and Dissemination (CRD) guidance for systematic reviews in healthcare and the Cochrane Collaboration's Handbook for Systematic Reviews of Interventions informed the review's methodology.11 ,12 The structure and content were informed by the preferred reporting items for systematic reviews and meta-analyses (PRISMA) guidelines.13 A protocol has been registered and published on the PROSPERO database (ref no: CRD42016050677).
Search strategy
Information sources
The following electronic databases were searched from September 2015 to January 2016 with the last search on 7 January 2016:
Cochrane Central Register of Controlled Trials and Cochrane Database of Systematic Reviews
PubMed (1980 onwards)
EMBASE (1980 onwards)
CINAHL (1981 onwards)
AMED (1985 onwards)
After initial broad scoping searches, the search terms outlined in table 1 were used to perform a focused systematic search. The population search was carried out first, followed by the intervention search. The search was carried out with the advice of the University of Warwick specialist librarian. Hand searching of references, ‘cited by’ and PubMed-related articles link searches were also carried out.
Search strategy
Inclusion/exclusion criteria
Inclusion and exclusion criteria are outlined in table 2.
Inclusion and exclusion criteria
Study selection
Duplicate articles were removed. Titles and abstracts were screened, followed by examination of the full text. Articles were assessed for inclusion independently by three reviewers (SM, KB and AM).
Data management
Two reviewers extracted relevant data to an Excel spreadsheet (AM and KB), which was independently checked for accuracy and detail by SM. The team discussed any disagreements.
Data synthesis
The included studies were compared and contrasted using a data extraction table. There were no comparable statistics and therefore a systematic narrative synthesis14 was undertaken, identifying cross-cutting themes from each study. The narrative was reviewed at intervals by LS, our patient and public involvement coauthor, with feedback provided on relevance to family experience and by JD for intellectual content.
Results
Study selection
Seven hundred and seventy relevant articles were identified. Seven hundred and fifty-five were excluded after title and abstract screening and the removal of duplicates, leaving 15 articles. Three of these were conference abstracts of ongoing studies which were not available as full-text articles or as unpublished studies from the authors and were therefore excluded. After applying the inclusion and exclusion criteria to the remaining 12 articles, 4 were excluded because they did not concern SPPC services with specialist medical support, leaving 8 articles. This process is shown in figure 1. Study characteristics are summarised in table 3.
Study characteristics
Preferred reporting items for systematic reviews and meta-analyses (PRISMA) flow diagram.
Study location
Included studies were from the USA,15–18 the UK,19 Germany,20 Australia21 and Canada.22
Study quality
The studies represented a heterogeneous body of evidence;13 seven were retrospective studies;15–19 ,21 ,22 three were surveys of bereaved parents,15 ,17 ,22 one was an epidemiological study19 and three were medical notes reviews,16 ,18 ,21 one of which included an economic analysis.18 There was one prospective longitudinal survey.20 There were no randomised-controlled trials or systematic reviews. All had clear aims and used appropriate methodology and approached the ethical issues. All acknowledged the limitations in their study design and recruitment strategies, and data were collected in a way that would address the research aims. All gave clear descriptions of their data analysis, results and findings.23
All had been published since 2012 and were carried out within single institutions or services. The largest study in terms of patient numbers was an epidemiological study, which looked at data regarding 2508 CYP but was limited by missing data items.19 The notes review studies examined the care of a total of 611 CYP.16 ,18 ,21 Three studies concerned only CYP with cancer.15 ,19 ,22 The other five studies concerned services for CYP with non-malignant conditions as well as those with cancer.16–18 ,20 ,21
Four studies made use of questionnaires with caregivers;15 ,17 ,20 ,22 three were surveys of bereaved parents.15 ,17 ,22 The total number of bereaved parents included in these studies was 200. Time since bereavement ranged from 7 months to over 4 years.15 ,17 ,22 Response rates for postal surveys of a total number of bereaved parents in a time period were 65/192 (37%)17 and 60/166 (36%).15 A response rate of 75/140 (75%) was achieved where eligibility criteria were applied.22 The highest response rate for a questionnaire survey was 93% (40/43), with the questionnaire administered face to face with family members at the time they were receiving care from the SPPC services.20 This study also attempted the assessment of children by self-report but due to young age and clinical condition this was possible with only three CYP.20
Key themes
Four key themes about how SPPC services can impact on CYP and their families were identified. These related to:
Quality of life
Symptom control
Place of care
Family support
Quality of life
The studies provide evidence that SPPC services contribute to improving the quality of life of CYP and family through emotional support, care planning and help with medical decision making,16 ,17 ,20 ,21 as well as through the management of distressing physical symptoms.17 ,21 Improved satisfaction with care was reported once SPPC services were involved.16 ,20 One study reported that CYP who were in contact with a SPPC “had more fun and [were] more likely to have an experience which added meaning to their life”15 than those who were not.
Symptom control
Pain and symptom management was described as one of the main reasons for referral to SPPC.21 Improvements in children's symptom control with the involvement of the SPPC team were reported by parents retrospectively.15 ,20 With the involvement of SPPC services, more care, including symptom management, was delivered in the home environment,15 alongside other support for caregivers and practical support.20
Place of care
Three studies provided evidence to suggest that referral to SPPC is associated with fewer admissions to hospital19 ,20 or a reduced length of stay.18 The involvement of SPPC services was associated with care planning discussions and considering a preferred location of death.16 ,22 More CYP died at home with SPPC involvement than not.15 ,20 Differences in terms of both diagnosis and geographical location of the family home contributed to the location of death. In one study, CYP with a cancer diagnosis were more likely to die at home if they lived in a rural location; CYP with non-malignant disease were more likely to die in a tertiary hospital.21 However, there was evidence that ‘goals of care’ discussions tended to occur relatively late, with the median time before death that this discussion took place being 16 days.16
Family experience
There were several areas in which SPPC intervention was reported as contributing to an improvement in family members' quality of life.20 ,22 Access to services 24 hours a day, 7 days/week was valued.20 Perception of psychological support and support for carrying out ‘day-to-day activities’ increased, and there was a decrease in anxiety and depression among parents.20 SPPC teams provided support with medical decision making,16 ,17 including discussions about resuscitation,16 ,22 help with communication between family members, including with their child, and with other healthcare teams.17 ,20 ,22 The SPPC team played an important role in educating parents about both the process of death and aspects of the medical system.17
Low referral rates to SPPC services were described19 and the average length of time that a child was under the care of SPPC varied from 20 days to over 1 year.16 ,18 ,21 ,22 Feedback from families included a desire that SPPC was involved earlier on in the course of a child's illness.17
Discussion
This review set out to investigate the current evidence regarding the distinct benefits to CYP and their families of care provided by SPPC, defined as a palliative care service supported by a physician with specialist training.
A small number of studies met the inclusion criteria, all of which had been published within the last 5 years. All of these were single-centre studies with relatively small patient numbers and rank low in the hierarchy of evidence due to their methodological limitations.12 This is a well-recognised problem in palliative care research, due to clinical and ethical challenges, and brings into question the value of systematic reviews in this subject area.24 Specific methodological challenges exist around gatekeeping of participants by clinicians,25 which was described as a barrier to recruitment in one study.22 In surveys, families were found to be unreachable by phone or post, did not respond or were ineligible to participate due to language barriers.15 ,17 ,20 ,22 Where families did respond, they are likely to have been particularly motivated to participate, and therefore the survey findings may not be generalisable to a more diverse population of families. One study tried to collect the views of CYP, but found this was not possible.20
Benefits of SPPC
Despite the limited quality of the evidence, there are cross-cutting themes from the eight studies suggesting that SPPC may enable improved quality of life for CYP and parents, improved symptom control, has an impact on the place of care and an increased likelihood that a preferred place of death is achieved.16 ,17 ,20–22
Service delivery
The review is timely as the specialty of paediatric palliative medicine further develops, with international standards, specifications and recommendations.8–10 ,26 Systematic consideration of the available evidence to support the development of services and policy is necessary, particularly as the number of CYP with LLCs and LTCs rises.
There is ongoing emphasis on place of death as an outcome measure in palliative care, despite a limited evidence base to support this.27 ‘Choice’ in end-of-life care is frequently highlighted in policy,28 and families desire the option of care being provided at home.29 This review suggests an association between referral to a SPPC team and opportunity to first express preferences for goals of care and location of death and then achieve it,15 ,16 ,22 although there was some evidence that this occurred relatively late in the course of illness.16 Key factors that enable these discussions have been described as continuous relationships, time for open, honest conversations and the provision of symptom control.15 ,20 ,29 ,30
Although adequate control of symptoms was not always perceived by parents,15 ,17 there was evidence to suggest that more effective symptom control could be delivered in the home environment with the involvement of an SPPC team.15 Further research into symptom management in CYP, including the use of medications and routes of administration, and how this can be delivered in both community and inpatient care settings, is an important focus for SPPC.
SPPC service design
What cannot be ascertained from the available evidence is which elements of SPPC services are associated with the benefits described, the mechanisms by which these benefits occur or the impact of the presence of a specialty-trained physician. This review looked specifically at services with a specialty-trained physician and excluded studies of any other model of care. However, nurse-led paediatric palliative care services and children's hospice services have also been shown to benefit CYP and their families, particularly in terms of place of care,31–34 coordination of care35 and family support.36 Research to compare more clearly the different types of services, and how they can effectively work together, would be valuable. Further research to investigate the most effective services for children with differing LLCs would also be of value given the wide variation in disease trajectories, family need and outcomes.3
The benefits of a specialist physician in a service have been broadly described as advanced clinical expertise and academic, educational and strategic leadership,37 all of which are important in SPPC as the specialty develops. Securing funding to develop both specialty training and new consultant posts presents a major challenge and will require clear business cases. Future innovation and development of the SPPC workforce and the implementation of new policies, including National Institute for Health and Care Excellence (NICE) guidance,26 should be accompanied by robust plans for evaluation.
This review identified only one study which made reference to the value of parental input into the development of SPPC services.17 Codesign of services with CYP and families28 ,38 and work to address possible reasons for low referral rates to SPPC, such as negative perceptions of palliative care among families39 and healthcare professionals,40 ,41 would be highly relevant.
Conclusion
Future service development recommendations should address the need for accessible and sustainable SPPC services for all CYP who need them. However, as this review demonstrates, there is limited evidence to inform policy guidance within the overall provision of paediatric palliative care. In the context of a growing number of CYP and families who could benefit from SPPC, there is a need for further research, innovation and debate. Robust evaluation of services, care models and professional roles, as well as research to understand the mechanisms by which benefits are delivered to CYP and families, is necessary. These are key considerations for those who are leading the development of SPPC and for service commissioners.
References
Footnotes
Contributors SM and JD designed the study. SM, AM and KB retrieved and analysed the data and drafted the article. AM and KB undertook these elements of the review to fulfil the requirements of the Selected Student Component 2 module of the MBChB course at Warwick University. JD assisted with interpretation of the data. LS and JD revised the article critically for clarity and intellectual content. All authors have approved this version for submission. All authors agree to be accountable for all aspects of the work in ensuring that questions related to the accuracy or integrity of any part of the work are appropriately investigated and resolved.
Funding SM is funded by a National Institute for Health Research Doctoral Research Fellowship (DRF-2014-07-065).
Disclaimer This article presents independent research funded in part by the National Institute for Health Research (NIHR). The views expressed are those of the authors and not necessarily those of the National Health Service, the NIHR or the Department of Health.
Competing interests None declared.
Provenance and peer review Not commissioned; externally peer reviewed.
Data sharing statement A systematic review protocol has been published on PROSPERO (ref no: CRD42016050677).