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Applications of artificial intelligence and the challenges in health technology assessment: a scoping review and framework with a focus on economic dimensions.
Background: Health Technology Assessment (HTA) is a crucial tool for evaluating the worth and roles of health technologies, and providing evidence-based guidance for their adoption and use. Artificial intelligence (AI) can enhance HTA processes by improving data collection, analysis, and decision-making. This study aims to explore the opportunities and challenges of utilizing artificial intelligence (AI) in health technology assessment (HTA), with a specific focus on economic dimensions. By leveraging AI's capabilities, this research examines how innovative tools and methods can optimize economic evaluation frameworks and enhance decision-making processes within the HTA context. Methods: This study adopted Arksey and O'Malley's scoping review framework and conducted a systematic search in PubMed, Scopus, and Web of Science databases. It examined the benefits and challenges of AI integration into HTA, with a focus on economic dimensions. Findings: AI significantly enhances HTA outcomes by driving methodological advancements, improving utility, and fostering healthcare innovation. It enables comprehensive assessments through robust data systems and databases. However, ethical considerations such as biases, transparency, and accountability emphasize the need for deliberate planning and policymaking to ensure responsible integration within the HTA framework. Conclusion: AI applications in HTA have significant potential to enhance health outcomes and decision-making processes. However, the development of robust data management strategies and regulatory frameworks is essential to ensure effective and ethical implementation. Future research should prioritize the establishment of comprehensive frameworks for AI integration, fostering collaboration among stakeholders, and improving data quality and accessibility on an ongoing basis.
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Systematic literature review of cost-effectiveness analyses of adult 15- and 20-valent pneumococcal vaccines.
Background: The economic and public health benefits of adult pneumococcal vaccines vary across countries due to different epidemiology and costs. We systematically reviewed and summarized findings and assumptions of cost-effectiveness analyses (CEA) of the recently introduced 15- and 20-valent pneumococcal conjugate vaccines (PCV15 and PCV20) in adults. Methods: We performed a systematic search for CEA studies of PCV15 and/or PCV20 versus existing strategies via PubMed, EMBASE, CEA Registry, EconLit, HTA Database, and NITAG resource center through April 23, 2024. Study characteristics, methods, assumptions, and findings were extracted independently by two reviewers; quality was assessed using ECOBIAS. Results were synthesized qualitatively to summarize key attributes and conclusions. Results: Of 137 identified records, 26 studies were included; the majority (24/26) concerned high-income countries. All employed static Markov-type models comparing higher-valent PCVs used alone or in combination with 23-valent pneumococcal polysaccharide vaccine (PPSV23) to current recommendations (PPSV23 alone, PCV13 alone, PCV13 + PPSV23, no vaccination). Most studies (22/26) concluded PCV20 used alone was cost-saving (dominant) or cost-effective compared to other adult pneumococcal strategies (PPSV23 alone, PCV13 ± PPSV23, PCV15 ± PPSV23, or no vaccination). PCVs were generally assumed to have serotype-specific effectiveness equal to PCV13 efficacy in the pivotal trial, though four studies used estimates from a Delphi panel; protection was assumed to last between 10 and 20 years. PPSV23 was assumed to have lower effectiveness against non-bacteremic pneumonia and shorter duration of protection. Herd effects from higher-valent PCVs in childhood (12/26), serotype replacement (2/26), or both (1/26) were included in half (13/26) of studies, which attenuated adult vaccine impact. Most studies were assessed as low risk of bias; five abstracts did not provide sufficient information for assessment. Conclusion: Current evidence indicates that 20-valent PCV used alone is likely to be cost-effective or dominate other adult pneumococcal strategies. Future research is needed to address remaining uncertainties in assumptions and to support evidence-based policymaking.
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Economic Evaluations of Chimeric Antigen Receptor T-Cell Therapies for Hematologic and Solid Malignancies: A Systematic Review
Objectives: This study aimed to systematically review evidence on the cost-effectiveness of chimeric antigen receptor T-cell (CAR-T) therapies for patients with cancer. Methods: Electronic databases were searched in October 2022 and updated in September 2023. Systematic reviews, health technology assessments, and economic evaluations that compared costs and effects of CAR-T therapy in patients with cancer were included. Two reviewers independently screened studies, extracted data, synthesized results, and critically appraised studies using the Philips checklist. Cost data were presented in 2022 US dollars. Results: Our search yielded 1809 records, 47 of which were included. Most of included studies were cost-utility analysis, published between 2018 and 2023, and conducted in the United States. Tisagenlecleucel, axicabtagene ciloleucel, idecabtagene vicleucel, ciltacabtagene autoleucel, lisocabtagene maraleucel, brexucabtagene autoleucel, and relmacabtagene autoleucel were compared with various standard of care chemotherapies. The incremental cost-effectiveness ratio (ICER) for CAR-T therapies ranged from $9424 to $4 124 105 per quality-adjusted life-year (QALY) in adults and from $20 784 to $243 177 per QALY in pediatric patients. Incremental costeffectiveness ratios were found to improve over longer time horizons or when an earlier cure point was assumed. Most studies failed to meet the Philips checklist due to a lack of head-to- head comparisons and uncertainty surrounding CAR-T costs and curative effects. Conclusions: CAR-T therapies were more expensive and generated more QALYs than comparators, but their cost-effectiveness was uncertain and dependent on patient population, cancer type, and model assumptions. This highlights the need for more nuanced economic evaluations and continued research to better understand the value of CAR-T therapies in diverse patient populations.
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Are intersectoral costs considered in economic evaluations of interventions relating to sexually transmitted infections (STIs)? A systematic review.
Background/objective: Sexually transmitted infections (STIs) not only have an impact on the health sector but also the private resources of those affected, their families and other sectors of society (i.e. labour, education). This study aimed to i) review and identify economic evaluations of interventions relating to STIs, which aimed to include a societal perspective; ii) analyse the intersectoral costs (i.e. costs broader than healthcare) included; iii) categorise these costs by sector; and iv) assess the impact of intersectoral costs on the overall study results. Methods: Seven databases were searched: MEDLINE (PubMed), EMBASE (Ovid), Web of Science, CINAHL, PsycINFO, EconLit and NHS EED. Key search terms included terms for economic evaluation, STIs and specific infections. This review considered trial- and model-based economic evaluations conducted in an OECD member country. Studies were included that assessed intersectoral costs. Intersectoral costs were extracted and categorised by sector using Drummond's cost classification scheme (i.e. patient/family, productivity, costs in other sectors). A narrative synthesis was performed. Results: Twenty-nine studies qualified for data extraction and narrative synthesis. Twenty-eight studies applied a societal perspective of which 8 additionally adopted a healthcare or payer perspective, or both. One study used a modified payer perspective. The following sectors were identified: patient/family, informal care, paid labour (productivity), non-paid opportunity costs, education, and consumption. Patient/family costs were captured in 11 studies and included patient time, travel expenses, out-of-pocket costs and premature burial costs. Informal caregiver support (non-family) and unpaid help by family/friends was captured in three studies. Paid labour losses were assessed in all but three studies. Three studies also captured the costs and inability to perform non-paid work. Educational costs and future non-health consumption costs were each captured in one study. The inclusion of intersectoral costs resulted in more favourable cost estimates. Conclusions: This systematic review suggests that economic evaluations of interventions relating to STIs that adopt a societal perspective tend to be limited in scope. There is an urgent need for economic evaluations to be more comprehensive in order to allow policy/decision-makers to make better-informed decisions.
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Economic Evaluation of Cognitive Behavioral Therapy for Depression: A Systematic Review
Objectives: This study aimed to conduct a systematic review of cost-utility studies of internet-based and face-to-face cognitive behavioral therapy (CBT) for depression from childhood to adulthood and to examine their reporting and methodological quality. Methods: A structured search for cost-utility studies concerning CBT for depression was performed in 7 comprehensive databases from their inception to July 2020. Two reviewers independently screened the literature, abstracted data, and assessed quality using the Consolidated Health Economic Evaluation Reporting Standards and Quality of Health Economic Studies checklists. The primary outcome was the incremental cost-effectiveness ratio (ICER) across all studies. To make a relevant comparison of the ICERs across the identified studies, cost data were inflated to the year 2020 and converted into US dollars. Results: Thirty-eight studies were included in this review, of which 26 studies (68%) were deemed of high methodological quality and 12 studies (32%) of fair quality. Despite differences in study designs and settings, the conclusions of most included studies for adult depression were general agreement; they showed that face-to-face CBT monotherapy or combination therapy compared with antidepressants and usual care for adult depression were cost-effective from the societal, health system, or payer perspective (ICER $241 212.4/quality-adjusted life-year [QALY] to $33 032.47/QALY, time horizon 12-60 months). Internet-based CBT regardless of guided or unguided also has a significant cost-effectiveness advantage (ICER $37 717.52/QALY to $73 841.34/QALY, time horizon 3-36 months). In addition, CBT was cost-effective in preventing depression (ICER $23 932.07/QALY to $26 092.02/QALY, time horizon 9-60 months). Nevertheless, the evidence for the cost-effectiveness of CBT for children and adolescents was still ambiguous. Conclusions: Fair or high-quality evidence showed that CBT monotherapy or combination therapy for adult depression was cost-effective; whether CBT-related therapy was cost-effective for children and adolescents depression remains inconclusive.
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More work is needed on cost-utility analyses of robotic-assisted surgery
Objective To comprehensively analyze the cost-utility of robotic surgery in clinical practice and to investigate the reporting and methodological quality of the related evidence. Methods Data on cost-utility analyses (CUAs) of robotic surgery were collected in seven electronic databases from the inception to July 2021. The quality of the included studies was assessed using the CHEERs and QHES checklists. A systematic review was performed with the incremental cost-effectiveness ratio as the outcome of interest. Results Thirty-one CUAs of robotic surgery were eligible. Overall, the identified CUAs were fair to high quality, and 63% of the CUAs ranked the cost-utility of robotic surgery as “favored,” 32% categorized as “reject,” and the remaining 5% ranked as “unclear.” Although a high heterogeneity was present in terms of the study design among the included CUAs, most studies (81.25%) consistently found that robotic surgery was more cost-utility than open surgery for prostatectomy (ICER: $6905.31/QALY to $26240.75/QALY; time horizon: 10 years or lifetime), colectomy (dominated by robotic surgery; time horizon: 1 year), knee arthroplasty (ICER: $1134.22/QALY to $1232.27/QALY; time horizon: lifetime), gastrectomy (dominated by robotic surgery; time horizon: 1 year), spine surgery (ICER: $17707.27/QALY; time horizon: 1 year), and cystectomy (ICER: $3154.46/QALY; time horizon: 3 months). However, inconsistent evidence was found for the cost-utility of robotic surgery versus laparoscopic surgery and (chemo)radiotherapy. Conclusions Fair or high-quality evidence indicated that robotic surgery is more cost-utility than open surgery, while it remains inconclusive whether robotic surgery is more cost-utility than laparoscopic surgery and (chemo)radiotherapy. Thus, an additional evaluation is required.
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Cost of Utilising Maternal Health Services in Low- and Middle-Income Countries: A Systematic Review.
Background: Cost is a major barrier to maternal health service utilisation for many women in low- and middle-income countries (LMICs). However, comparable evidence of the available cost data in these countries is limited. We conducted a systematic review and comparative analysis of costs of utilising maternal health services in these settings. Methods: We searched peer-reviewed and grey literature databases for articles reporting cost of utilising maternal health services in LMICs published post-2000. All retrieved records were screened and articles meeting the inclusion criteria selected. Quality assessment was performed using the relevant cost-specific criteria of the Consolidated Health Economic Evaluation Reporting Standards (CHEERS) checklist. To guarantee comparability, disaggregated costs data were inflated to 2019 US dollar equivalents. Total adjusted costs and cost drivers associated with utilising each service were systematically compared. Where heterogeneity in methods or non-disaggregated costs was observed, narrative synthesis was used to summarise findings. Results: Thirty-six studies met our inclusion criteria. Many of the studies costed multiple services. However, the most frequently costed services were utilisation of normal vaginal delivery (22 studies), caesarean delivery (13), and antenatal care (ANC) (10). The least costed services were post-natal care (PNC) and post-abortion care (PAC) (5 each). Studies used varied methods for data collection and analysis and their quality ranged from low to high with most assessed as average or high. Generally, across all included studies, cost of utilisation progressively increased from ANC and PNC to delivery and PAC, and from public to private providers. Medicines and diagnostics were main cost drivers for ANC and PNC while cost drivers were variable for delivery. Women experienced financial burden of utilising maternal health services and also had to pay some unofficial costs to access care, even where formal exemptions existed. Conclusion: Consensus regarding approach for costing maternal health services will help to improve their relevance for supporting policy-making towards achieving universal health coverage. If indeed the post-2015 mission of the global community is to "leave no one behind," then we need to ensure that women and their families are not facing unnecessary and unaffordable costs that could potentially tip them into poverty.
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Cost-Effectiveness of Endovascular Versus Open Repair of Abdominal Aortic Aneurysm: A Systematic Review.
Purpose: Abdominal aortic aneurysm (AAA) is a life-threatening condition which, in the absence of increasing diameter or rupture, often remains asymptomatic, and a diameter greater than 5.5 cm requires elective surgical repair. This study aimed to evaluate the cost-effectiveness of endovascular repair (EVAR) versus open surgical repair (OSR) in patients with AAA through a systematic review of published health economics studies. Methods: Using a systematic review method, an electronic search was conducted for cost-effectiveness studies published on AAA (both in English and Persian) on PubMed, Embase, ISI/Web of Science (WoS), SCOPUS, Global Health databases, and the national databases of Iran from 1990 to 2020 including the keywords "cost-effectiveness", "endovascular", "open surgical", and "abdominal aortic aneurysms". The quality of the studies was assessed using the Quality of Health Economic Studies (QHES) checklist. Results: In total, 958 studies were found, of which 16 were eligible for further study. All studies were conducted in developed countries, and quality-adjusted life years (QALY) and life years (LY) were used to measure the outcomes. According to the QHES checklist, most studies were of good quality. In European countries and Canada, EVAR has not been cost-effective, while most studies in the United States regard this technique as a cost-effective intervention. For example, incremental cost-effectiveness ratio (ICER) values ranged from $14,252.12 to $34,446.37 per QALY in the USA, while ICER was €116,600.40 per QALY in Portugal. Conclusion: According to the results, the EVAR technique has been more cost-effective than OSR for high-risk patients, but the need for continuous follow-up, increased costs, and re-intervention over the long term and for low-risk patients has reduced the cost-effectiveness of this method. As the health systems vary among different countries (i.e. quality of care, cost of devices, etc.), and due to the heterogeneity of studies in terms of the follow-up period, time horizon, and threshold, all of which are inherent features of economic evaluation, generalizing the results should be done with much caution, and policymaking must be based on national evidence.
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Economic evaluation of the utilization of telemedicine for patients with cardiovascular disease: a systematic review.
Cardiovascular disease is one of the major causes of mortality in the world, with high human and financial burdens on communities. Telemedicine is a tool for providing services for patients that are difficult to access or in need of immediate care. The aim of this study was to systematically review economic evaluation studies that compared telemedicine with usual care for cardiovascular patients. A systematic review was conducted according to Preferred Reporting Items for Systematic Reviews and Meta-Analyses guidelines and related articles published up to December 2018 were searched in different databases (PubMed, Embase, SCOPUS, Global Health, Google Scholar, Magiran, SID). The articles were selected based on inclusion and exclusion criteria. Consolidated health economic evaluation reporting standards statement checklist was used to qualitatively evaluate the papers. Overall, 20 articles were included in the study. The studies used quality-adjusted life years to measure outcomes. The highest and lowest values of Incremental Cost-effectiveness Ratio were $515,082 and $2099 that had been reported in the UK and New Zealand, respectively. Most of the items of the checklist were reported and the quality of most of the studies was excellent and very good. According to the results, telemedicine improves the clinical outcomes and results in considerable saving in costs. Utilizing telemedicine concurrent with the usual care for service delivery is more cost-effective. The reviewed studies had been conducted in high-income countries, hence, it is essential to be cautious when generalizing the results and applying them in health systems policymaking.
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Economic evaluations of pharmacist-led medication review in outpatients with hypertension, type 2 diabetes mellitus, and dyslipidaemia: a systematic review.
Objectives: To evaluate the health economics evidence based on randomized controlled trials of pharmacist-led medication review in pharmacotherapy managed cardiovascular disease risk factors, specifically, hypertension, type-2 diabetes mellitus and dyslipidaemia in ambulatory settings and to provide recommendations for future evaluations. Methods: A systematic review was carried out according to the Cochrane Handbook for Systematic Reviews. PubMed (Medline), Scopus, Web of Science, National Health System Economic Evaluation Database (NHS EED), Cochrane Library, and Econlit were searched and screened by two independent authors. Incremental cost-effectiveness ratio was the main outcome. Risk of bias was assessed with the Effective Practice and Organisation of Care tool by the Cochrane Collaboration. Economic evaluation quality was assessed with the he Consensus Health Economic Criteria list (CHEC list). Results: 5636 records were found, and 174 were retrieved for full-text review yielding 11 articles. Eight articles deemed the intervention as cost effective and two as dominant. Two cost-utility analyses were performed yielding ICERs of $612.7 and $59.8 per QALY. Four articles were considered to perform a high-quality economic evaluation and four had a low risk of bias. Future economic evaluations should consider cost-utility analysis, to describe usual care thoroughly, and use time horizons that capture the effect of cardiovascular disease prevention, a societal perspective and uncertainty analysis. Conclusion: Pharmacist-led medication review has proven to be cost effective in various studies in different settings. Policy decision makers are advised to undertake local economic evaluations reflecting the gaps observed in this systematic review and published literature. If this is not possible, a transferability assessment should be conducted.
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Economic evaluations in community aged care: A systematic review
BACKGROUND: This paper reports the methods and findings from a systematic review of economic evaluations conducted in the community aged care sector between 2000 and 2016. METHODS: Online databases searched were PubMed, Medline, Scopus, and web of science, CINAHL and informit. Studies were included if they 1) were full economic evaluations that compared both the costs and outcomes of two or more interventions 2) in study population of people aged 65 years and over 3) dependent older people living in the community 4) alternatives being compared were care models or service delivery interventions in the community aged care sector (a group of programs that have been established as a support system to allow older people to remain living in their own homes for as long as possible, as an alternative to institutional or residential care) and 5) published in the English language between 2000 and November 2016. RESULTS: Eleven studies reporting upon economic evaluations of service delivery interventions in community aged care were identified; the majority of which were undertaken in Europe. Critical appraisal of the identified studies highlighted the methodological rigour in these evaluations. CONCLUSION: This systematic review highlights the paucity of economic evaluation studies conducted to date in the community aged care sector. The findings highlight the importance of cost utility analysis methodology as it allows for a uniform outcome measure, that facilitates the comparison of different interventions. In addition, multi-attribute utility measures that represent those quality of life domains that are most important to older people should be used and attention must be paid to the inclusion of informal care costs and outcomes as this is a key resource in community aged care service delivery.
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Economic evaluation of dental sealants: A systematic literature review
Objectives: To systematically review the literature on economic evaluations of dental sealants and examine the costs and effectiveness of caries prevention using sealants. Methods: Of 21 full-text articles examined, a total of 13 were included in this study. These studies are grouped by the type of intervention as follows: (i) sealants compared with no sealants; (ii) sealants compared with other forms of caries prevention; (iii) resin-based sealants compared with glass-ionomer sealants; (iv) different sealing strategies in primary teeth; (v) different sealing strategies in permanent teeth; and (vi) sealants based on school- or clinic-based setting of delivery. All currency is reported in constant 2010 US$. Results: Cost-effectiveness analyses differed due to varying study designs, assumptions, sealant delivery settings, outcomes, caries risk assessment and study durations. Findings varied on the cost-effectiveness of sealants compared with other caries-preventive strategies. Under the assumption of equal caries risk, always sealing primary molars appeared to be the most effective strategy, whereas risk-based sealing was the optimal strategy with differing caries risk. Studies that assessed sealing strategies in permanent teeth reported that risk-based sealing was more cost-effective than not sealing, but they differed on the cost-effectiveness of risk-based seal compared with non-risk-based seal. Sealants delivered in school settings had mixed results on costs but were as equally effective as sealants delivered in private practices. Conclusions: The cost-effectiveness of sealants is dependent on the conditions of delivery. The list of cost-effectiveness ratios for each intervention can support policy makers to estimate expected returns on their investments in dental sealants.
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Economic evaluations of ergonomic interventions preventing work-related musculoskeletal disorders: a systematic review of organizational-level interventions.
Background: Work-related musculoskeletal disorders (WMSD) represent a major public health problem and economic burden to employers, workers and health insurance systems. This systematic review had two objectives: (1) to analyze the cost-benefit results of organizational-level ergonomic workplace-based interventions aimed at preventing WMSD, (2) to explore factors related to the implementation process of these interventions (obstacles and facilitating factors) in order to identify whether economic results may be due to a successful or unsuccessful implementation. Methods: Systematic review. Studies were searched in eight electronic databases and in reference lists of included studies. Companion papers were identified through backward and forward citation tracking. A quality assessment tool was developed following guidelines available in the literature. An integration of quantitative economic results and qualitative implementation data was conducted following an explanatory sequential design. Results: Out of 189 records, nine studies met selection criteria and were included in our review. Out of nine included studies, grouped into four types of interventions, seven yielded positive economic results, one produced a negative result and one mixed results (negative cost-effectiveness and positive net benefit). However, the level of evidence was limited for the four types of interventions given the quality and the limited number of studies identified. Our review shows that among the nine included studies, negative and mixed economic results were observed when the dose delivered and received by participants was low, when the support from top and/or middle management was limited either due to limited participation of supervisors in training sessions or a lack of financial resources and when adequacy of intervention to workers' needs was low. In studies where economic results were positive, implementation data showed strong support from supervisors and a high rate of employee participation. Conclusion: Studies investigating the determinants of financial outcomes of prevention related to implementation process are very seldom. We recommend that in future research economic evaluation should include information on the implementation process in order to permit the interpretation of economic results and enhance the generalizability of results. This is also necessary for knowledge transfer and utilization of research results for prevention-oriented decision-making in occupational health and safety.
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Economic evaluations of interventions designed to prevent mental disorders: A systematic review
AIM:The prevention of mental disorders is a growing field and there are interventions that have been demonstrated to prevent some disorders, particularly depression, from developing. The aim of the current study is to update two existing reviews of the cost-effectiveness studies of preventive interventions for mental disorders in order to determine whether such interventions are good value-for-money. METHODS: A search was undertaken in Medline, PsycInfo and Econlit. The search was limited to articles published in English covering the period from 2010 to September 2013. Inclusion criteria for the review comprised comparative economic evaluations of interventions designed to prevent mental disorders. RESULTS: Ten new economic evaluations have been published since 2010, more than doubling the numbers of economic evaluations of preventive interventions for mental disorders published prior to 2010. Using the Consolidated Health Economic Evaluation Reporting Standards (CHEERS) checklist, the majority of studies were of a good standard and used cost-utility frameworks. CONCLUSIONS: Indicated types of interventions for the prevention of depression and anxiety appeared to be particularly good value-for-money and most of these studies were modelled evaluations. Unfortunately, many such interventions are still not routinely provided. Future trials of preventive interventions for mental disorders need to include robust economic evaluations so that the economic impact of such interventions from the individual study participant perspective can be determined.
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The cost-effectiveness of patient decision aids: A systematic review
The Affordable Care Act includes provisions to encourage patient-centered care through the use of shared decision making (SDM) and patient decision aids (PtDA). PtDAs are tools that can help encourage SDM by providing information about competing treatment options and elucidating patients values and preferences. Implementing PtDAs into routine practice may incur additional costs through training or increases in physician time. Prominent commentaries have proposed that these costs might be offset if patients choose less expensive options than their providers. However, the cost-effectiveness of PtDAs to date is unclear. The aim of this study was to review the economic evidence from PtDA trials. Our search identified 5347 articles, with 29 included following full-text review. Only one economic evaluation of a PtDA has been completed, which found a PtDA to be cost-saving in women with menorrhagia. Other studies included in the review indicated that PtDAs will likely increase up-front costs, but in some contexts may reduce short-term costs by reducing the uptake of invasive treatments, such as elective surgery. Few studies comprehensively captured long-term costs or measured benefits in a manner conducive to economic evaluation (QALYs or general health utilities). Our review suggests that policy makers currently have insufficient economic evidence to appropriately consider their investments in PtDAs
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Economic evaluation of cystic fibrosis screening: A review of the literature
Objectives To critically examine the economic evidence regarding cystic fibrosis (CF) carrier screening and to understand issues relating to the transferability of international findings to any national context for policy decisions. Methods A systematic literature search identified 14 studies (out of 29 economic studies on CF) focusing on preconception or prenatal screening between 1990 and 2006. These studies were then assessed against international benchmarks on conducting and reporting of economic evaluations, costing methodology used and focusing on the transferability of the evidence to national contexts. Results The primary outcome measures varied considerably between studies and there was considerable ambiguity and variation on how costs were estimated. The Incremental Cost Effectiveness Ratio (ICER) and net savings, for preconception and prenatal screening were inconsistent and varied significantly, even after adjusting for timing and exchange rates. Differences in screening participation rates, reproductive choices, test sensitivity, cost of test and lifetime cost of care make up a large part of the ICER variations. Conclusion The heterogeneity in study design, model inputs and reporting of economic evaluations of CF carrier screening makes comparability and transferability across countries and even within countries difficult. This reinforces the need to assess any technology within the relevant context, and to not simply generalize from reported studies. In turn, this adds to the complex task of making efficient resource allocation decisions in the area of CF carrier screening. Our evaluation adds weight to the calls for revisiting the way economic studies are conducted and reported.
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