Introduction
The overarching aim for this study was:
To use a mixed methods approach including quantitative analyses of national audit data and qualitative approaches to gather information from key individuals, in order to establish an evidence based and realistic guideline for community based surveillance of fragile infants with congenital heart disease.153
With the specific study objective:
To combine the data and information acquired to generate the evidence based protocol or guideline for surveillance of infants with CHD, including the ‘who’, ‘when’ and ‘how’ this should best be delivered. The ultimate objective is to produce a workable and effective follow-up surveillance protocol for infants discharged into the community after cardiac surgery, with appropriate targeting of higher risk patients and consideration of measures that will be acceptable and useful to parents and community based health care professionals. Intervention development will include consideration of measures of success.153
This chapter discusses the development of our suggestions for service improvement in respect of the postdischarge management of fragile infants with CHD, which represent the main deliverable of the project, fulfilling the overarching study aim and objective. The evidence that was collected and analysed in each phase of the study over the course of 2 years, including the two systematic reviews of the literature (objective 1), the national audit data that generated a risk model for our outcomes of interest and CART risk groups (objective 2), and our interviews and other qualitative explorations (objective 3), were presented and discussed in an iterative fashion to a multidisciplinary group, over the course of the entire study. Each of these elements of research evidence informed and fed into subsequent stages of work, ultimately generating our final suggestions for service improvement (objective 4) and informing the implications for future practice and research that are discussed in this chapter.
Methods
To the end of fulfilling the above stated aim and objective, an expert advisory group to IHS was established to review the emerging evidence generated from the systematic reviews, quantitative analyses and qualitative analyses relating to the outcomes and discharge/postdischarge management of neonates and infants undergoing intervention for CHD and propose candidate suggestions for improving services based on that evidence. This multidisciplinary group comprised professionals (with a diverse skill set) from the three tertiary cardiac centres involved in the study (K Brown, K Bull, D Barron, R Franklin and P Daubeney), representatives from primary (S Hull) and secondary care (N Barnes), patient group representatives (R Simbodyal) and academics from the disciplines of psychology (J Tregay and J Wray), statistics (D Ridout), epidemiology (R Knowles) and operational research (S Crowe).
The group met on five occasions (each lasting 2–3 hours) between March 2013 and June 2014 to consider:
the results of mixed-methods research regarding UK service provision and outcomes specifically in this patient population as discussed in
Chapters 2 and
3 (systematic reviews),
Chapters 4 and
5 (quantitative analyses of national audit data) and
Chapters 6–
9 (qualitative analyses of OF data, as well as family, professional and charity helpline views)
quality improvement initiatives in related areas such as the discharge of premature and high-risk infants.
In July 2014, these candidate suggestions and a summary of the evidence were shared at a facilitated workshop with parents of infants who had undergone surgery for CHD and subsequently either died after discharge or required emergency readmission to intensive care (10 parents representing five babies). Their views on the acceptability of the candidate suggestions and additional ones were captured (notes from the meeting are included in full in Appendix 9).
This phase of the project drew upon the skills and input of an operational researcher trained in quality improvement methods (S Crowe) who was funded by the Health Foundation and joined the study team with the express purpose of bringing operational research expertise to bear upon this phase of the study.
Establishing the final set of suggestions for health care
A working group comprising selected members of the expert advisory group and invited additional representatives from the community and charitable sector was convened to assess the draft suggestions for health care and propose a final set for endorsement by the IHS expert advisory group (see Appendix 10 for the working group’s terms of reference and list of members). A facilitated all-day workshop was held in September 2014 (tape-recorded and with live minutes), in which the group was tasked with:
reviewing the draft suggestions for health care: assessing the feasibility and acceptability of each for service improvement
assessing the set of draft suggestions for health care as a whole within the context of patient risk groups and targeting these (setting priorities)
agreeing a final set of suggestions for health-care improvement to circulate among the IHS expert advisory group for comments and endorsement.
On the second point, the working group explicitly considered both the size and nature of the risk associated with each of the patient groups identified in our analysis of national audit data reported in Chapter 4 (summarised in ). The groups are characterised by patient risk factors and stratified by occurrence of outcome 2 (death or emergency readmission to paediatric intensive care within the first year post discharge from infant cardiac surgery). Any decision about the types of interventions supported for different patient groups will have implications for resource use. Given the limited resources available for providing these services, the working group therefore agreed that it was important to consider the relative size of the patient groups alongside their relative risks and the nature of those risks when thinking about how limited resources might be targeted most effectively.
Results
The proposals for service improvement that represent the main output of the project are set out in . The table is structured to show the aspects of the service that might benefit from improvement alongside the source of the evidence on which each individual statement is based, as collected over the various phases of the study in the left-hand column. The proposals put forward by the intervention development group, which are linked to each individual area for improvement, are listed in the right-hand column. The elements of the patient journey that are covered within are training and information for families predischarge, discharge and transferring to non-specialist services, medical follow-up services, non-medical support, provision of patient information, accessing support when a baby is sick and knowledge gaps, weak links and poor communication between HPs.
Endorsed suggestions for service improvements
It is worth noting that the original overarching study objective was focused on the community-based surveillance of fragile infants with CHD, and as listed in , this area has been covered in detail in our suggestions for service improvement (see the sections on medical follow-up services, non-medical support, provision of patient information and accessing support when a baby is sick). In undertaking the iterative series of reviews of the emerging evidence from the study it became apparent that optimal community-based surveillance of these infants necessitates further inter-related improvements to additional elements of the patient journey (see sections on training and information for families predischarge and discharge and transferring to non-specialist services). Hence our focus widened somewhat to incorporate aspects of the patient journey that start in the tertiary centre (including training of families and communication between professionals in different sectors) and these additional aspects are discussed in detail in .
Many of the service improvements proposed and refined by the working group were suggested for all infants as they were considered to have lower resource implications and/or to be important for everyone, irrespective of their risk (e.g. a structured discharge document or standardised training). However, interventions considered particularly beneficial for certain patient groups were suggested selectively (e.g. multidisciplinary care teams for children with long term complex needs in addition to their primary cardiac diagnosis). In addition, given that over half of all adverse events occur in the 21% of patients in risk groups 1–4 (see ), suggestions regarding some of the potentially higher resource aspects of service provision were prioritised for these high-risk patient groups (e.g. discharge home via the local hospital). Finally, on the basis of qualitative evidence (Chapters 7 and 8), suggesting that problems in accessing support can be exacerbated for families who face cultural or language barriers or have learning difficulties, and on the basis that National Audit data presented in Chapter 4 indicates that more complex heart defects, deprivation and higher-risk medical conditions such as neurodevelopmental problems are over-represented in particular ethnic minority groups, some suggestions were prioritised for these groups (e.g. offering and supporting family buddying).
The service challenges and barriers identified during the course of the IHS are listed below, accompanied by the suggestions for service improvements endorsed by the IHS expert advisory group.
Discussion
Over the course of the IHS a set of evidence-informed guidelines were developed for managing the discharge and postdischarge care of infants undergoing major cardiac intervention, which were directly aimed at addressing challenges in this previously neglected section of the care pathway. The development of this guideline was, indeed, the overarching aim of the study, and as stated above in the Introduction section of this chapter, formed the basis of study objective 4. In the completion of study objectives 1–3 and the gathering of multimethods research evidence that generated information regarding patient risk characteristics and the challenges encountered in accessing and providing services for this patient population presented in Chapters 2–9, it became clear that the guideline would need to incorporate a series of comments and suggestions related to discharge and postdischarge management, including community-based surveillance of fragile infants with heart disease. These guidelines, which are presented in full in , represent the final output of an evidence-informed, multistakeholder process, and go beyond the original aims of our study in that they are somewhat broader in scope. This was necessary in order to address comprehensively all of the evidence that came to light over the course of the programme of research.
Strengths and limitations
A limitation of our research project was that the development of optimal guidelines for services across sectors is challenging, not least because there is often limited or disparate evidence that is difficult to synthesise (for example with no established methods equivalent to the meta-analysis of randomised controlled trials), and inevitably involves an element of subjectivity. In this context, our study had two key strengths. First, developing and applying a systematic and transparent process for synthesising and incorporating a broad range of available evidence covering multiple aspects of the problem enhanced the richness and breadth of the guidelines. For example, the qualitative evidence was very useful in specifying what the problems in services were and how they might be improved, and the quantitative evidence enabled prioritisation of patient groups according to their risk. Second, representatives from across the entire patient pathway critiqued the feasibility and acceptability of the recommendations, and the needs of service users remained of central importance through incorporating findings from an OF and interviews, views captured in a family workshop and involving a parent representative on the expert group.
Implications for practice
Our proposed guidelines for services are of direct relevance to all health-care professionals caring for infants with CHD including GPs, community nurses, HVs, secondary care paediatricians and clinicians in specialist surgical centres, as well as patients, their families and support groups. The proposals will resonate with clinicians, patient families and user groups from other geographical areas when later postdischarge CHD deaths have been reported as a concern in certain diagnostic groups, for example Germany55 and the USA.26
As discussed in more detail in , there is a need for reduced variability and improved overall standards in respect of the training and information for families with infants affected by CHD predischarge, the process for discharge and transfer of infants with CHD to non-specialist services and their medical follow-up services, the non-medical support for patient families, the provision of patient information, the access to support when a baby is sick, and in reducing knowledge gaps and strengthening communication between HPs. It is likely that such interventions could have a material impact in terms of reducing postdischarge deaths and readmissions to intensive care, as well as improving the perceptions and experiences of services by patients’ families.
There is a role for the relevant professional groups (e.g. the British Congenital Cardiac Association and the PEC Special Interest Group), research funders, the national audit body and service users in taking forward certain aspects that require further research and development (see Recommendations for research). CHD services in the UK are currently under national review by NHS England and the findings and conclusions reported in this article fed into the review’s public consultation on the care standards and service specification to be used in commissioning specialist CHD services (see Appendix 11).
Suggestions for audit metrics
The evidence and suggestions from the IHS would support monitoring of the process and outcome measures, in order to increase the focus on this neglected phase of the patient journey and hence, it is hoped, to drive up standards of service provision. One potential option for such monitoring is the Quality Dashboard, which is guided by the Clinical Reference Group. Please note, however, that while the IHS team suggests the monitoring of out-of-hospital outcomes was highlighted as important within the IHS data, the analytical steps and processes required to do this are beyond the scope of the IHS team and would need to be considered further by, for example, the NCHDA steering committee.
The IHS findings would first need to be disseminated more widely within the community and agreement reached about the steps required for implementing the recommendations; for example, a number of metrics relate to standardised documents/protocols that would need to be developed and appropriately piloted beforehand. Others require further consideration as to how the information could be collected and/or aggregated. Furthermore, some findings may be considered more appropriate for local monitoring for improvement purposes, rather than for inclusion on the national Quality Dashboard.
The IHS therefore suggests that the feasibility and appropriateness of including any or all of the following metrics from Box 30 on the Quality Dashboard should be revisited in the next 1 to 2 years and, in the meantime, steps taken to develop these areas.
Metrics proposed for use in audit Emergency unplanned readmissions to PICU within 1 year following infant heart surgery (for all infants).
Patient-reported experience measures
The IHS supports the monitoring of patient-reported experience measures alongside the process and outcome measures in Box 30, for example adopting a similar approach to the specialist Quality Dashboards (e.g. heart transplant and ECMO) that include three questions relating to patient-reported experience measures. This area would require further development work.
The importance of joined-up service specifications/commissioning across specialist, local and community services
We emphasise that the IHS findings demonstrate the need and potential for improvements across the entire patient journey spanning community, primary, secondary and specialist services. This would require service specifications and commissioning to be addressed not only for the specialist services commissioned by the Congenital Heart Services clinical reference group, but for all of these sectors. The IHS’s evidence of weak links across sectors and poor communication between different HPs further suggests the need for joined-up service specifications and models of commissioning across the whole patient journey, including local and community settings.
Recommendations for research
The study highlighted areas where further specific research is needed, these being as follows.
Home monitoring programmes
The systematic review (see Chapter 3) revealed that data on the effectiveness of HMPs are limited: most of the data originate from the USA (where health systems differ), with only one study available from Germany; in addition, the data’s effectiveness is not completely certain as control patients were based on historic data, which means that resultant biases should be considered, such as the role of other changes in the patient journey apart from HMPs contributing to improvements. The qualitative data in our study from the family and professional interviews reported in Chapters 7 and 8 indicated that although many informants thought that HMPs were beneficial because they helped everyone to follow consistent standards and increased awareness across various HP groups of high-risk patients, some parents found HMPs burdensome. The studies in the systematic review did not contain user viewpoints, so we cannot ascertain what these were. Furthermore, current practice across the UK in respect of HMP protocols varies widely (see Chapter 8). Overall, the studies in the systematic review support the hypothesis that HMP are effective (with some remaining doubts and caveats), and our study findings support the use of HMP for HLHS, UVH and PA patients with shunt dependence. However, further national consensus building and research is required to establish the optimal protocol and components of HMP incorporating specific consideration of user views and cultural or language barriers before taking these forward. Additional work is also needed to review the inclusion criteria for HMP, particularly when these necessitate additional patients being monitored over current practice, including an assessment of resource implications. As HMP are implemented more uniformly within UK centres, it will be vital to collect prospective information about effectiveness.
Development and evaluation of proposed tools for improvement
Additional health-care evaluation is required of the best format, applications and effectiveness of the proposed traffic-light tool to detect early warning signs of deterioration, as well as health-care evaluation of the proposed structured discharge document, discharge checklist and step-down care. Cultural and language barriers should form part of this evaluation.
Metrics for national audit
The study working group has clearly endorsed the need to include metrics for national audit and benchmarking, which go beyond 30-day mortality rates and address the need to monitor postdischarge outcomes for infant CHD. Given the availability of life status tracking to the national audit (NCHDA) and the possibility being open for record linkage to occur between NCHDA and PICANet as a means to underpin future audit (this would enable the audit of postdischarge emergency readmissions to PICU), this is technically feasible. That said, a series of analytical steps would need to be undertaken for this to occur, which are not insignificant.
Another important issue to consider, which is likely to require further research, is adjustment for case mix when monitoring postdischarge outcomes. The first step that the working group proposed, of auditing postdischarge interstage deaths in infants with HLHS, UVH and PA, incorporates case mix adjustment, as these diagnoses represent a high-risk group as determined by our analyses. However, we note that the risks of adverse outcome differ even between HLHS (odds ratio of outcome 1 : 7.4) and UVH–PA (odds ratio of outcome 1 : 4.4). In this example, the national distribution of children with HLHS across specialist centres is uneven in the UK with certain centres taking care of the majority of patients with HLHS. If outcome is audited for HLHS together with UVH and PA, then there is the potential for an unfairly negative assessment of outcome in a centre with a large number of patients that have HLHS. Hence, finally, we note that further analytical and statistical steps are required in order to adequately address case mix complexity, building upon the work we have set out in Chapter 4, such that these outcomes and further postdischarge outcome measures proposed above for future audit, may effectively form part of the portfolio of metrics subjected to national audit.
