scholarly journals Holistic services for people with advanced disease and chronic or refractory breathlessness: a mixed-methods evidence synthesis

2019 ◽  
Vol 7 (22) ◽  
pp. 1-104 ◽  
Author(s):  
Matthew Maddocks ◽  
Lisa Jane Brighton ◽  
Morag Farquhar ◽  
Sara Booth ◽  
Sophie Miller ◽  
...  

Background Breathlessness is a common and distressing symptom of many advanced diseases, affecting around 2 million people in the UK. Breathlessness increases with disease progression and often becomes chronic or refractory. Breathlessness-triggered services that integrate holistic assessment and specialist palliative care input as part of a multiprofessional approach have been developed for this group, offering tailored interventions to support self-management and reduce distress. Objectives The aim was to synthesise evidence on holistic breathlessness services for people with advanced disease and chronic or refractory breathlessness. The objectives were to describe the structure, organisation and delivery of services, determine clinical effectiveness, cost-effectiveness and acceptability, identify predictors of treatment response, and elicit stakeholders’ evidence-based priorities for clinical practice, policy and research. Design The mixed-methods evidence synthesis comprised three components: (1) a systematic review to determine the clinical effectiveness, cost-effectiveness and acceptability of holistic breathlessness services; (2) a secondary analysis of pooled individual data from three trials to determine predictors of clinical response; and (3) a transparent expert consultation (TEC), comprising a stakeholder workshop and an online consensus survey, to identify stakeholders’ priorities. Results Thirty-seven papers reporting on 18 holistic breathlessness services were included in the systematic review. Most studies enrolled people with thoracic cancer, were delivered over 4–6 weeks, and included breathing training, relaxation techniques and psychological support. Meta-analysis demonstrated significant reductions in the Numeric Rating Scale (NRS) distress due to breathlessness, significant reductions in the Hospital Anxiety and Depressions Scale (HADS) depression scores, and non-significant reductions in the Chronic Respiratory Disease Questionnaire (CRQ) mastery and HADS anxiety, favouring the intervention. Recipients valued education, self-management interventions, and expertise of the staff in breathlessness and person-centred care. Evidence for cost-effectiveness was limited and inconclusive. The responder analysis (n = 259) revealed baseline CRQ mastery and NRS distress to be strong predictors of the response to breathlessness services assessed by these same measures, and no significant influence from baseline breathlessness intensity, patient diagnosis, lung function, health status, anxiety or depression. The TEC elicited 34 priorities from stakeholders. Seven priorities received high agreement and consensus, reflecting stakeholders’ (n = 74) views that services should be person-centred and multiprofessional, share their breathlessness management skills with others, and recognise the roles and support needs of informal carers. Limitations The evidence synthesis draws predominantly from UK services and may not be generalisable to other settings. Some meta-analyses were restricted by reporting biases and statistical heterogeneity. Conclusions Despite heterogeneity in composition and delivery, holistic breathlessness services are highly valued by recipients and can lead to significant improvements in the distress caused by breathlessness and depression. Outcomes of improved mastery and reduced distress caused by breathlessness are not influenced by patient diagnosis, lung function or health status. Stakeholders highlighted the need for improved access to person-centred, multiprofessional breathlessness services and support for informal carers. Future work Our research suggests that key therapeutic components of holistic breathlessness services be considered in clinical practice and models of delivery and educational strategies to address stakeholders’ priorities tested. Study registration This study is registered as PROSPERO CRD42017057508. Funding The National Institute for Health Research (NIHR) Health Services and Delivery programme. Matthew Maddocks, Wei Gao and Irene J Higginson are supported by the NIHR Collaboration for Leadership in Applied Health Research and Care (CLAHRC) South London; Matthew Maddocks is supported by a NIHR Career Development Fellowship (CDF-2017-009), William D-C Man is supported by the NIHR CLAHRC Northwest London and Irene J Higginson holds a NIHR Emeritus Senior Investigator Award.

2021 ◽  
Vol 21 (1) ◽  
Author(s):  
Saowalak Turongkaravee ◽  
Jiraphun Jittikoon ◽  
Onwipa Rochanathimoke ◽  
Kathleen Boyd ◽  
Olivia Wu ◽  
...  

Abstract Background Genetic testing has potential roles in identifying whether an individual would have risk of adverse drug reactions (ADRs) from a particular medicine. Robust cost-effectiveness results on genetic testing would be useful for clinical practice and policy decision-making on allocating resources effectively. This study aimed to update a systematic review on economic evaluations of pharmacogenetic testing to prevent ADRs and critically appraise the quality of reporting and sources of evidence for model input parameters. Methods We searched studies through Medline via PubMed, Scopus and CRD’s NHS Economic Evaluation up to October 2019. Studies investigating polymorphism-based pharmacogenetic testing, which guided drug therapies to prevent ADRs, using economic evaluation methods were included. Two reviewers independently performed data extraction and assessed the quality of reporting using the Consolidated Health Economic Evaluation Reporting Standards (CHEERS) guidelines and the quality of data sources using the hierarchy of evidence developed by Cooper et al. Results Fifty-nine economic evaluations of pharmacogenetic testing to avoid drug-induced ADRs were found between 2002 and 2018. Cost-utility and cost-effectiveness analyses were the most common methods of economic evaluation of pharmacogenetic testing. Most studies complied with the CHEERS checklist, except for single study-based economic evaluations which did not report uncertainty analysis (78%). There was a lack of high-quality evidence not only for estimating the clinical effectiveness of pharmacogenetic testing, but also baseline clinical data. About 14% of the studies obtained clinical effectiveness data of testing from a meta-analysis of case-control studies with direct comparison, which was not listed in the hierarchy of evidence used. Conclusions Our review suggested that future single study-based economic evaluations of pharmacogenetic testing should report uncertainty analysis, as this could significantly affect the robustness of economic evaluation results. A specific ranking system for the quality of evidence is needed for the economic evaluation of pharmacogenetic testing of ADRs. Differences in parameters, methods and outcomes across studies, as well as population-level and system-level differences, may lead to the difficulty of comparing cost-effectiveness results across countries.


Author(s):  
Mandana Zanganeh ◽  
Peymane Adab ◽  
Bai Li ◽  
Emma Frew

Many suggested policy interventions for childhood and adolescent obesity have costs and effects that fall outside the health care sector. These cross-sectorial costs and consequences have implications for how economic evaluation is applied and although previous systematic reviews have provided a summary of cost-effectiveness, very few have conducted a review of methods applied. We undertook this comprehensive review of economic evaluations, appraising the methods used, assessing the quality of the economic evaluations, and summarising cost-effectiveness. Nine electronic databases were searched for full-economic evaluation studies published between January 2001 and April 2017 with no language or country restrictions. 39 economic evaluation studies were reviewed and quality assessed. Almost all the studies were from Western countries and methods were found to vary by country, setting and type of intervention. The majority, particularly “behavioural and policy” preventive interventions, were cost-effective, even cost-saving. Only four interventions were not cost effective. This systematic review suggests that economic evaluation of obesity interventions is an expanding area of research. However, methodological heterogeneity makes evidence synthesis challenging. Whilst upstream interventions show promise, an expanded and consistent approach to evaluate cost-effectiveness is needed to capture health and non-health costs and consequences.


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