T
he aim of this research project was to evaluate the clinical effectiveness and cost-effectiveness of interventions aiming to improve the mental health of CYP with LTCs and to explore the factors that may enhance, or limit, the beneficial delivery of such interventions. To address these aims we conducted two systematic reviews, one focused on quantitative studies and one on the qualitative evidence related to relevant interventions. We then brought together findings from each review in an overarching synthesis.As part of the project we consulted with a range of end-users about preliminary findings, strengthening the main arguments and developing key implications on the basis of these activities. A CYPAG was formed and met four times during the project to provide input into methodological decisions, to consult on preliminary findings and to help prepare dissemination materials. This final chapter summarises the findings of each review and the overarching synthesis and describes the strengths and limitations of the work conducted, outlining the implications for practice and recommendations for further research.
Summary of findings
Summary of review 1
In review 1 (see Chapter 2), we synthesised 25 RCTs (31 articles) that assessed the clinical effectiveness of interventions aiming to improve the mental health of CYP with LTCs and elevated symptoms of mental ill health. These studies evaluated 11 types of intervention provided to CYP with 12 different types of LTC.
We extracted 269 outcome measures from the 31 articles. Although the primary focus of the review was the impact of interventions on the mental health of CYP, all secondary outcomes were synthesised to evaluate any benefits of interventions on other aspects of young people’s lives, including key aspects of individual and family functioning. As many different measures of similar underlying constructs were reported, we categorised them into 28 categories. Seventeen categories related to CYP mental health;
the rest related to‘other outcomes’.
The greatest volume of research focused on the clinical effectiveness of CBT, which was evaluated in seven studies (10 articles). These studies provide tentative evidence that CBT-based interventions could be beneficial for the mental health of CYP with IBD, chronic pain, epilepsy and persistent functional somatic complaints, but not T1DM. The majority of the CBT interventions had content that was adapted to the needs of the LTC of CYP in the sample. In addition, four parenting programme interventions were tested in three studies. Group play therapy interventions were also assessed in three studies. Other intervention types (palliative care, relaxation, SMT, emotional intelligence training, massage therapy, biofeedback, resistance training and music therapy) were reported in only one or two studies.
We calculated effect sizes for each study outcome whenever data allowed. Trials were typically small, so effect sizes across the included studies were characterised by wide CIs. Therefore, the evidence for particular interventions used with similar samples of CYP is very limited. There were relatively few opportunities to meta-analyse studies with similar intervention, comparator, LTC and outcomes. No more than two studies shared particular combinations of these study characteristics and could be meta-analysed.
Study quality of the relatively small amount of research to date is poor, with 19 out of the 25 studies having at least four separate indicators of potential bias. Blinding of assessors and evaluation of adherence, fidelity and compliance were often neglected, and reporting of randomisation methods and allocation concealment was largely unclear. Fewer than half of the studies included a follow-up assessment, with only one-half of those occurring beyond 6 months post intervention. The quality of future research in the field should therefore be improved. Although findings from review 1 are tentative, they support previous research.
A systematic review by Bennett et al.82of psychological interventions for mental health disorders in CYP with chronic conditions also reported preliminary evidence that CBT interventions may be beneficial.
DOI: 10.3310/hta23220 HEALTH TECHNOLOGY ASSESSMENT 2019 VOL. 23 NO. 22
© Queen’s Printer and Controller of HMSO 2019. This work was produced by Moore et al. under the terms of a commissioning contract issued by the Secretary of State for Health and Social Care. This issue may be freely reproduced for the purposes of private research and study and extracts (or indeed, the full report) may be included in professional journals provided that suitable acknowledgement is made and the reproduction is not associated with any form of advertising. Applications for commercial reproduction should be addressed to: NIHR Journals Library, National Institute for Health Research, Evaluation, Trials and Studies Coordinating Centre, Alpha House, University of Southampton Science Park, Southampton SO16 7NS, UK.
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Review 1 is the first systematic review to consider all interventions for elevated symptoms of mental ill health in CYP with LTCs.
National Institute for Health and Care Excellence guidelines recommend CBT for CYP with depression.54 James et al.70published a Cochrane systematic review of CBT for childhood anxiety disorders in CYP without comorbid LTCs, reporting a suggestion that CBT is a more effective treatment than treatment as usual or active controls. Given our own findings, there is tentative evidence that these recommendations may hold for CYP with LTCs. Review 1 does not explain why CBT has promising effects, or whether or not and how to adapt it to CYP with LTCs for the best effect. Future research should seek to explore these questions.
The findings of review 1 point to the need for large, high-quality RCTs with consistency in intervention, design and outcome reporting. Rigorous testing of interventions across a range of conditions and locations, including fidelity and process evaluation measures, would aid comparison of the components of different interventions. It would be particularly useful to examine whether or not intervention types that show some promise (e.g. CBT) are clinically effective across a range of LTCs, including whether or not (and how) they should be adapted to specific LTCs. Although we included studies of interventions that aimed to improve mental health, the majority also targeted other outcomes, particularly LTC symptoms. Therefore, future research might consider treatment integrating both physical and mental health care and its effects across a wider range of outcomes, rather than focus primarily on mental health in terms of population, intervention and outcome.
Summary of review 2
In review 2 (see Chapter 3), 57 studies were reviewed, which included qualitative data collection and analysis on the experience of, and attitude towards, interventions aiming to improve the mental health and well-being of CYP with LTCs. These studies evaluated a more diverse range of interventions (n = 21) than that seen in review 1. The most frequently seen LTC in the 57 included studies was cancer (n = 14).
HIV-positive intervention recipients were the focus of eight studies, whereas a further 10 studies included CYP with a mix of LTCs, something that was not seen in review 1. Included studies commonly explored the perceptions and experiences of interventions that aimed to improve aspects of mental health and well-being such as coping (n = 26), self-esteem (n = 13) and emotional support (n = 11) rather than targeting a mental health disorder, for example depression (n = 1) or anxiety (n = 8). Interventions also often aimed to improve symptoms related to the LTC (n = 35) and social skills (n = 16). Included studies represented views from a range of different participants involved in the delivery and receipt of relevant interventions.
We used metaethnography to synthesise the 57 included studies (60 articles). The synthesis revealed five main constructs:‘a therapeutic foundation’, ‘social support’, ‘a hopeful alternative’, ‘resilience’ and
‘getting in and staying in’. A number of themes contributed to each construct. We developed a line of argument (a model explaining how themes and constructs related to each other) that offers an explanatory model of the experience of receiving and delivering interventions to improve the mental health of CYP with LTCs (see Figure 18). The model describes the process by which CYP with LTCs access and maintain engagement with a relevant intervention aiming to improve mental health and well-being. Some
interventions can help CYP to acquire a sense of hope for the future and increase their resilience.
Interventions are often perceived to be effective when they offer participants a safe space and social support. The relationship between these constructs is not fully defined, as other factors may have an impact on the experience of interventions and participants’ attitudes.
A young person’s sense of resilience, hope and social support as created through taking part in an intervention may help them and encourage them to maintain their engagement with the intervention.
Their experience may also encourage CYP and their carers to build positive relationships and use these relationships and the skills learnt during interventions to develop techniques to manage emotions, physical symptoms and other relationships. Interventions that can respond flexibly to the physical and mental health
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needs of the CYP for whom it is intended may also help keep the CYP engaged with the intervention over time. Such flexibility, along with the acquisition of new skills, may help build and maintain the effects of interventions beyond the intervention setting. There is a tension between (1) the perceived need for an intervention to be flexible in content and delivery to respond to individual needs and also provide a sense of being unconstrained and (2) the need for boundaries. Evidence suggests that, although CYP welcome the opportunity to escape from the worries and constraints associated with living with a LTC in everyday life, they require certain limits on behaviour, discussion topics and physical activity to experience an intervention as safe. The balance required to ensure that these two concepts are incorporated into an intervention may represent a challenge to those who design and deliver it.
The quality of included articles was generally good. At least 10 high-quality articles contributed to each theme in the synthesis. However, general weaknesses included a failure to make the theoretical perspective of the author explicit, a failure to adequately describe the context or setting of qualitative research and a lack of clarity in the description of the intervention.
This is the first attempt to examine the experiences of participants in interventions that aim to improve the mental health and well-being of CYP with LTCs. However, our synthesis shares features with some previous literature. Concepts from Wallander et al.’s37disability-stress coping model of adjustment to chronic illness (e.g. problem solving and perceived competence) appear to be similar to our themes of
‘empowerment’ and ‘skills and knowledge’. Some of the specific resistance factors seen within Wallander et al.’s37model, including the role of improved family relationships, sense of competence and use of coping strategies, are also seen in our line of argument.
Our themes‘I am not alone’ and ‘therapeutic relationships’, which explore how CYP valued feelings of connectedness and belonging with peers and empathy/validation from intervention deliverers, fit with the findings of a systematic review of young people’s experience of mental health in chronic illness.45Our themes‘unconstrained’ and ‘a new normal’ are also reflected in findings from this previous review that CYP view their LTC as something that restricts participating in‘normal’ life and everyday activities and is a source of uncertainty both in the present and with regard to worries about their future.45The importance of social support, development of coping strategies and a sense of hope and acceptance are also
acknowledged in both our review 2 and the previous review.45
There was only one study in which qualitative data collection and analysis occurred alongside a RCT, which was also included in review 1;119there is a need for this type of mixed-methods process evaluation conducted according to best practice guidance to improve our understanding of how interventions are experienced and how this may link to effectiveness.241
Summary of the overarching synthesis
The overarching synthesis (see Chapter 4) integrated the findings from review 1 and review 2. A deductive approach was used, whereby questions based on the emerging findings of each review were generated and used to interrogate the other review for information that could potentially inform the findings or explain gaps in the literature. Despite the different review questions, methods of synthesis and types of interventions seen across the two systematic reviews, the overarching synthesis allowed us to raise a number of tentative implications.
Nine categories emerged from the analysis:
1. degree of overlap between the two reviews 2. availability of up-to-date, good-quality research 3. what works for whom
4. adaptations to interventions and flexibility 5. accessibility and delivery of interventions 6. stress and coping
DOI: 10.3310/hta23220 HEALTH TECHNOLOGY ASSESSMENT 2019 VOL. 23 NO. 22
© Queen’s Printer and Controller of HMSO 2019. This work was produced by Moore et al. under the terms of a commissioning contract issued by the Secretary of State for Health and Social Care. This issue may be freely reproduced for the purposes of private research and study and extracts (or indeed, the full report) may be included in professional journals provided that suitable acknowledgement is made and the reproduction is not associated with any form of advertising. Applications for commercial reproduction should be addressed to: NIHR Journals Library, National Institute for Health Research, Evaluation, Trials and Studies Coordinating Centre, Alpha House, University of Southampton Science Park, Southampton SO16 7NS, UK.
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7. working with family or peers 8. therapeutic relationships 9. holistic approach.
There is a limited amount of overlap between the reviews, particularly when focusing on interventions used for particular populations. Much of the research included in both reviews was published in the 10 years prior to the review, but there is a noticeable lack of studies conducted in the UK. Although there is some evidence of benefit for interventions in CYP with particular LTC populations, for example CBT in populations with IBD and music therapy for procedural distress in CYP with cancer, neither the quantitative nor qualitative evidence can explain why this may be or suggest how intervention effectiveness may vary between individuals.
The review work suggests that further evidence is needed to understand whether or not and how interventions should be adapted for the LTCs of CYP. The accessibility and familiarity of an intervention’s setting, use of technology and flexibility of delivery can have an impact on perceived effectiveness and may warrant consideration as to how such issues affect effectiveness in terms of mental health outcomes and wider functioning. There was evidence from both reviews that interventions that include family, particularly parents, may be beneficial. Relationships between CYP with LTCs and their therapists, and with their ill and healthy peers, seem important. Although both reviews include interventions that aim to improve mental health in some way, the majority of interventions also targeted other outcomes including LTC symptoms, social support, knowledge and self-management.
Strengths and limitations
There are a number of strengths of the project’s reviews, overarching synthesis and end-user involvement:
l Review 1 represents the first (to our knowledge) systematic review of RCTs evaluating interventions targeting mental health of CYP with LTCs. It was conducted and is reported in accordance with best practice guidelines.85,113
l We have used comprehensive search strategies and made extensive efforts to locate research outside academic databases.
l We have also broadened the scope of review 1 compared with previous attempts to synthesise this evidence base by including populations with elevated symptoms of mental ill health in samples as opposed to only symptoms of mental health disorders or diagnoses.
l For review 1, searches were not limited to English-language papers, date or study location, and authors were contacted for supplementary materials and missing data when necessary. Although we targeted papers reporting on interventions that primarily sought to improve CYP mental health, we included and synthesised all wider outcomes, therefore providing a fuller picture of the effects of interventions.
l Review 2 is the first attempt (to our knowledge) to synthesise research on the experience of those involved in the delivery and receipt of interventions that aim to improve the mental health of CYP with LTCs. A strength of the review is that studies included the views of a range people with an interest in such interventions, namely CYP, caregivers and intervention deliverers on interventions at different stages in development.
l The synthesis within review 2 considered a large number of papers in a time-effective manner and prioritised richer, interpretive findings, before using the descriptive studies to support or refute aspects of the synthesis.
l Our overarching synthesis brought together findings from both reviews. In doing so, we were able to highlight clearer implications for practice and gaps for further research.
l We consulted with a wide range of end-users about our preliminary findings and used feedback to refine our analysis and write-up of the final report.
l We convened a CYPAG that met at four points during the project. This group gave feedback on the use of terminology for the project, commented on preliminary findings, co-produced the Plain English summary and recorded audio for a podcast disseminating the work.
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We also acknowledge the following limitations, which predominantly stemmed from the primary research reviewed:
l The breadth of outcomes reported in review 1 presented a challenge for analysis and interpretation.
For example, depression was assessed using 14 different instruments across 14 different studies.
The breadth of measures used made comparability across samples challenging when different scales assessing the same construct were used, as the meaning of established cut-off points may differ across scales. When it was not possible to find an established cut-off score in the literature for measures used, the article was considered not to have met inclusion criteria, even though it is possible the sample may have had unreported elevated symptoms.
l There were insufficient studies to allow meaningful moderator analysis of intervention type and LTC population or assess publication bias.
l No economic evaluations related to the included trials or that otherwise fitted our inclusion criteria were located, which indicates a need for measures of cost-effectiveness to be incorporated in trials.
l No UK studies were included in review 1, therefore limiting the applicability of review findings to the UK context.
l There may be subjectivity in the categorisation of interventions. For instance, we may have grouped progressive muscle relaxation, heart rate variability biofeedback and massage together if there was an argument for these intervention targets or active ingredients being similar. Categorisation was on the basis of similar content and targets, meaning that several interventions could not be categorised with any others.
l Each study included in this review sampled CYP participants with particular LTCs, with the result that we do not have a clear sense regarding the applicability of particular interventions for improving mental health across a range of LTCs.
l Relevant interventions may have been excluded from the synthesis because of our inclusion criterion that the intervention had to be described as aiming to improve the mental health or well-being of CYP with a LTC.
l Owing to the potential impact of misinterpretation of abstract concepts following translation from other languages, only English-language articles were included in review 2. There was a wide range in the type of qualitative analysis used within the studies, which meant that the analyses conducted in individual studies were not always directly comparable. The categorisation of study findings and purposive sampling strategy as described in Chapter 3, Methods, Methods of analysis/synthesis, Data analysis and synthesis,
l Owing to the potential impact of misinterpretation of abstract concepts following translation from other languages, only English-language articles were included in review 2. There was a wide range in the type of qualitative analysis used within the studies, which meant that the analyses conducted in individual studies were not always directly comparable. The categorisation of study findings and purposive sampling strategy as described in Chapter 3, Methods, Methods of analysis/synthesis, Data analysis and synthesis,