Economic evaluations of nonpharmacological treatments for drug-resistant epilepsy: A systematic review.

This study was undertaken to systematically identify and critically appraise all published full economic evaluations assessing the cost-effectiveness of nonpharmacological interventions for patients with drug-resistant epilepsy. The Population, Intervention, Comparison, Outcome, Study criteria was used to design search strategies for the identification and selection of relevant studies. Literature search was performed using the MEDLINE (via PubMed), Embase, International Health Technology Assessment, National Institute for Health Research Economic Evaluation Database, and Cost-Effectiveness Analysis Registry databases to identify articles published between January 2000 and May 2023. Web of Science was additionally used to perform forward and backward referencing. Title, abstract, and full-text screening was performed by two independent researchers. The Consensus Health Economic Criteria (CHEC) checklist and Consolidated Health Economic Evaluation Reporting Standards (CHEERS) 2022 were applied for quality assessment. A total of 4470 studies were identified, of which 18 met our inclusion criteria. Twelve of the studies conducted model-based economic evaluation, and others were trial-based. Three studies showed that epilepsy surgery was cost-effective in adults, whereas this remained inconclusive for children (two positive, three negative). Three studies showed negative economic outcome for ketogenic diet in children. One of four studies showed positive results for self-management. For vagus nerve stimulation, one study showed positive results in adults and another one negative results in children. One recent study showed cost-effectiveness of responsive neurostimulation (RNS) in adults. Finally, one study showed promising but inconclusive results for deep brain stimulation (DBS). The mean scores for risk of bias assessment (based on CHEC) and for reporting quality (CHEERS 2022) were 95.8% and 80.5%, respectively. This review identified studies that assessed the cost-effectiveness of nonpharmacological treatments in both adults and children with drug-resistant epilepsy, suggesting that in adults, epilepsy surgery, vagus nerve stimulation, and RNS are cost-effective, and that DBS and self-management appear to be promising. In children, the cost-effectiveness of epilepsy surgery remains inconclusive. Finally, the use of ketogenic diet was shown not to be cost-effective. However, limited long-term data were available for newer interventions (i.e., ketogenic diet, DBS, and RNS).

Development of an Instrument for the Assessment of Health-Related Multi-sectoral Resource Use in Europe: The PECUNIA RUM.

BACKGROUND: Measuring objective resource-use quantities is important for generating valid cost estimates in economic evaluations. In the absence of acknowledged guidelines, measurement methods are often chosen based on practicality rather than methodological evidence. Furthermore, few resource-use measurement (RUM) instruments focus on the measurement of resource use in multiple societal sectors and their development process is rarely described. Thorn and colleagues proposed a stepwise approach to the development of RUM instruments, which has been used for developing cost questionnaires for specific trials. However, it remains unclear how this approach can be translated into practice and whether it is applicable to the development of generic self-reported RUM instruments and instruments measuring resource use in multiple sectors. This study provides a detailed description of the practical application of this stepwise approach to the development of a multi-sectoral RUM instrument developed within the ProgrammE in Costing, resource use measurement and outcome valuation for Use in multi-sectoral National and International health economic evaluAtions (PECUNIA) project. METHODS: For the development of the PECUNIA RUM, the methodological approach was based on best practice guidelines. The process included six steps, including the definition of the instrument attributes, identification of cost-driving elements in each sector, review of methodological literature and development of a harmonized cross-sectorial approach, development of questionnaire modules and their subsequent harmonization. RESULTS: The selected development approach was, overall, applicable to the development of the PECUNIA RUM. However, due to the complexity of the development of a multi-sectoral RUM instrument, additional steps such as establishing a uniform methodological basis, harmonization of questionnaire modules and involvement of a broader range of stakeholders (healthcare professionals, sector-specific experts, health economists) were needed. CONCLUSION: This is the first study that transparently describes the development process of a generic multi-sectoral RUM instrument in health economics and provides insights into the methodological aspects and overall validity of its development process.

Do Costs in the Education Sector Matter? A Systematic Literature Review of the Economic Impact of Psychosocial Problems on the Education Sector.

BACKGROUND: Psychosocial (e.g., anxiety or behavior) problems lead to costs not only in the healthcare sector but also in education and other sectors. As psychosocial problems develop during the critical period of establishing educational trajectories, education costs are particularly relevant in the context of psychosocial problems among children and adolescents. OBJECTIVES: This study aimed to gain insights into the methods used for the inclusion of education costs in health economics studies and into the proportion of the education costs in relation to the total costs associated with a condition or an intervention. METHODS: We systematically searched the PubMed, Embase, SSCI, CINAHL, PsycINFO, ERIC, and Econlit databases in August 2019 for economic evaluations of mental health, psychosocial and educational interventions, and cost-of-illness studies of mental, behavioral, and neurodevelopmental disorders conducted from a societal perspective in populations of children and adolescents. An additional search was conducted in February 2021 to update the review. RESULTS: In total, 49 articles were included in the analysis. The most common cost items were special education, school absenteeism, and various educational professionals (educational psychologist). A variety of methods were employed for the identification, measurement, and/or valuation of education costs. The proportion of education costs to the total costs of condition/intervention ranged from 0 to 67%, with the mean being 18.5%. DISCUSSION: Since education costs can constitute a significant proportion of the total costs of an intervention or condition, including them in health economics studies might be important in informing optimal resource allocation decisions. Although various methods are available for including education costs in health economics studies, further research is needed to develop evidence-based methods for producing comparable estimates.

Aspects and Challenges of Resource Use Measurement in Health Economics: Towards a Comprehensive Measurement Framework.

BACKGROUND: While the methods for conducting health economics research in general are improving, current guidelines provide limited guidance regarding resource use measurement (RUM). Consequently, a variety of methods exists, yet there is no overview of aspects to consider when deciding on the most appropriate RUM methodology. Therefore, this study aims to (1) identify and categorize existing knowledge regarding aspects of RUM, and (2) develop a framework that provides a comprehensive overview of methodological aspects regarding RUM. METHODS: Relevant articles were identified by enrolling a search string in six databases and handsearching the DIRUM database. Included articles were descriptively reviewed and served as input for a comprehensive framework. Health economics experts were involved during the process to establish the framework's face validity. RESULTS: Forty articles were included in the scoping review. The RUM framework consists of four methodological RUM domains: 'Whom to measure', addressing whom to ask and whom to measure; 'How to measure', addressing the different approaches of measurement; 'How often to measure', addressing recall period and measurement patterns; and 'Additional considerations', which covers additional aspects that are essential for further refining the methodologies for measurement. Evidence retrieved from the scoping review was categorized according to these domains. CONCLUSION: This study clustered the aspects of RUM methodology in health economics into a comprehensive framework. The results may guide health economists in their decision making regarding the selection of appropriate RUM methods and developing instruments for RUM. Furthermore, policy makers may use these findings to review study results from an evidence-based perspective.

The broader societal impacts of COVID-19 and the growing importance of capturing these in health economic analyses.

AbstractThe rapid spread of the current COVID-19 pandemic has affected societies worldwide, leading to excess mortality, long-lasting health consequences, strained healthcare systems, and additional strains and spillover effects on other sectors outside health (i.e., intersectoral costs and benefits). In this perspective piece, we demonstrate the broader societal impacts of COVID-19 on other sectors outside the health sector and the growing importance of capturing these in health economic analyses. These broader impacts include, for instance, the effects on the labor market and productivity, education, criminal justice, housing, consumption, and environment. The current pandemic highlights the importance of adopting a societal perspective to consider these broader impacts of public health issues and interventions and only omit these where it can be clearly justified as appropriate to do so. Furthermore, we explain how the COVID-19 pandemic exposed and exacerbated existing deep-rooted structural inequalities that contribute to the wider societal impacts of the pandemic.

The Relative Importance of Education and Criminal Justice Costs and Benefits in Economic Evaluations: A Best-Worst Scaling Experiment.

OBJECTIVES: Mental and behavioural disorders (MBDs) and interventions targeting MBDs lead to costs and cost savings in the healthcare sector, but also in other sectors. The latter are referred to as intersectoral costs and benefits (ICBs). Interventions targeting MBDs often lead to ICBs in the education and criminal justice sectors, yet these are rarely included in economic evaluations. This study aimed to investigate the attitudes held by health economists and health technology assessment experts towards education and criminal justice ICBs in economic evaluations and to quantify the relative importance of these ICBs in the context of MBDs. METHODS: An online survey containing open-ended questions and two best-worst scaling object case studies was conducted in order to prioritise a list of 20 education ICBs and 20 criminal justice ICBs. Mean relative importance scores for each ICB were generated using hierarchical Bayes analysis. RESULTS: Thirty-nine experts completed the survey. The majority of the respondents (68%) reported that ICBs were relevant, but only a few (32%) included them in economic evaluations. The most important education ICBs were "special education school attendance", "absenteeism from school", and "reduced school attainment". The most important criminal justice ICBs were "decreased chance of committing a crime as a consequence/effect of mental health programmes/interventions", "jail and prison expenditures", and "long-term pain and suffering of victims/victimisation". CONCLUSIONS: This study identified the most important education and criminal justice ICBs for economic evaluations of interventions targeting MBDs and suggests that it could be relevant to include these ICBs in economic evaluations.

Exploring the identification, validation, and categorization of the cost and benefits of criminal justice in mental health: the PECUNIA project.

BACKGROUND: Mental health disorders and their treatments produce significant costs and benefits in both healthcare and non-healthcare sectors. The latter are often referred to as intersectoral costs and benefits (ICBs). Little is known about healthcare-related ICBs in the criminal justice sector and how to include these in health economics research. OBJECTIVES: The triple aim of this study is (i) to identify healthcare-related ICBs in the criminal justice sector, (ii) to validate the list of healthcare-related ICBs in the criminal justice sector on a European level by sector-specific experts, and (iii) to classify the identified ICBs. METHODS: A scientific literature search in PubMed and an additional grey literature search, carried out in six European countries, were used to retrieve ICBs. In order to validate the international applicability of the ICBs, a survey was conducted with an international group of experts from the criminal justice sector. The list of criminal justice ICBs was categorized according to the PECUNIA conceptual framework. RESULTS: The full-text analysis of forty-five peer-reviewed journal articles and eleven grey literature sources resulted in a draft list of items. Input from the expert survey resulted in a final list of fourteen unique criminal justice ICBs, categorized according to the care atom. CONCLUSION: This study laid further foundations for the inclusion of important societal costs of mental health-related interventions within the criminal justice sector. More research is needed to facilitate the further and increased inclusion of ICBs in health economics research.

Intersectoral Costs and Benefits of Mental and Behavioural Disorders in the Education Sector: an Exploration of Costing Methods.

BACKGROUND: The inclusion of indirect spillover costs and benefits that occur in non-healthcare sectors of society is necessary to make optimal societal decisions when assessing the cost effectiveness of healthcare interventions. Education costs and benefits are relevant in the disease area of mental and behavioral disorders, but their inclusion in economic evaluations is largely neglected due to lack of methodological knowledge. AIM OF THE STUDY: This study aims to explore, using a scoping review, the identification, measurement, and valuation methods used to assess the impact of mental and behavioural disorders on education costs and benefits. METHODS: A scoping review was conducted to identify articles that were set in the education sector and assessed education costs and benefits. An adapted 5-step approach was used: (i) initating a scoping review; (ii) identifying component studies; (iii) data extraction; (iv) reporting results; (v) discussion and interpretation of findings. Results were summarized in a narrative synthesis per identification, measurement, and valuation method. RESULTS: 177 component articles were identified in the scoping review that reported 61 mutually exclusive education costs and benefits. The nomenclature used to describe the costs and benefits was poorly defined, heterogeneous in nature and largely context dependent. This was also reflected in the diverse number of measurement and valuation methods found in the component articles. DISCUSSION: This is the first study, which offers a classification of education costs and benefits and costing methods reported by studies set in the education sector. In conclusion, mental and behavioral disorders have a notable impact on a variety of different education costs and benefits. IMPLICATIONS FOR HEALTH POLICIES: The classification provided in the current study gives an indication of the wide-spread impact of mental and behavioral disorders on the education sector. Hence, the inclusion of relevant education costs and benefits in economic evaluations for mental and behavioral disorders is necessary to make optimal societal decisions. IMPLICATIONS FOR FURTHER RESEARCH: By exploring a new area of research from a sector-specific perspective, the current study adds to the existing intersectoral cost and benefit literature base. Future research should focus on standardizing costing methods in pharmacoeconomic guidelines and assessing the relative importance of individual education costs and benefits in economic evaluations for specific interventions and diseases.