scholarly journals Incorporating Cascade Effects of Genetic Testing in Economic Evaluation: A Scoping Review of Methodological Challenges

Children ◽  
2021 ◽  
Vol 8 (5) ◽  
pp. 346
Author(s):  
Alexandra Cernat ◽  
Robin Z. Hayeems ◽  
Lisa A. Prosser ◽  
Wendy J. Ungar

Cascade genetic testing is indicated for family members of individuals testing positive on a genetic test, and is particularly relevant for child health because of their vulnerability and the long-term health and economic implications. Cascade testing has patient- and health system-level implications; however cascade costs and health effects are not routinely considered in economic evaluation. The methodological challenges associated with incorporating cascade effects in economic evaluation require examination. The purpose of this scoping review was to identify published economic evaluations that considered cascade genetic testing. Citation databases were searched for English-language economic evaluations reporting on cascade genetic testing. Nineteen publications were included. In four, genetic testing was used to identify new index patients—cascade effects were also considered; thirteen assessed cascade genetic testing strategies for the identification of at-risk relatives; and two calculated the costs of cascade genetic testing as a secondary objective. Methodological challenges associated with incorporating cascade effects in economic evaluation are related to study design, costing, measurement and valuation of health outcomes, and modeling. As health economic studies may currently be underestimating both the cost and health benefits attributable to genetic technologies through omission of cascade effects, development of methods to address these difficulties is required.

Author(s):  
Alexandra Cernat ◽  
Robin Z. Hayeems ◽  
Wendy J. Ungar

AbstractCascade genetic testing is the identification of individuals at risk for a hereditary condition by genetic testing in relatives of people known to possess particular genetic variants. Cascade testing has health system implications, however cascade costs and health effects are not considered in health technology assessments (HTAs) that focus on costs and health consequences in individual patients. Cascade health service use must be better understood to be incorporated in HTA of emerging genetic tests for children. The purpose of this review was to characterise published research related to patterns and costs of cascade health service use by relatives of children with any condition diagnosed through genetic testing. To this end, a scoping literature review was conducted. Citation databases were searched for English-language papers reporting uptake, costs, downstream health service use, or cost-effectiveness of cascade investigations of relatives of children who receive a genetic diagnosis. Included publications were critically appraised, and findings were synthesised. Twenty publications were included. Sixteen had a paediatric proband population; four had a combined paediatric and adult proband population. Uptake of cascade testing varied across diseases, from 37% for cystic fibrosis, 39% to 65% for hypertrophic cardiomyopathy, and 90% for rare monogenic conditions. Two studies evaluated costs. It was concluded that cascade testing in the child-to-parent direction has been reported in a variety of diseases, and that understanding the scope of cascade testing will aid in the design and conduct of HTA of emerging genetic technologies to better inform funding and policy decisions.


Author(s):  
Silvia M.A.A. Evers ◽  
Carmen D. Dircksen

Introduction: Stakeholders are increasingly interested in the societal impact of psychosocial interventions in the youth sector, in terms of costs and quality of life, as well as in outcomes research. The aim of this broad consultation was to reach consensus regarding the steps to be undertaken to set a research agenda for the Netherlands Organisation for Health Research and Development (ZonMw) programme. Methods: The broad consultation consisted of an eight-step procedure, including the conceptualization of a consultation document consisting of a scoping review of (mainly) international opinion/methodological literature and an inventory of existing Dutch guidelines and manuals for economic evaluation, a written consultation procedure among a broad range of stakeholders, and a consultation meeting with these stakeholders. Results: In total 21 documents were included in the scoping review. A total of 24 stakeholders participated in the written consultation procedure and 14 stakeholders during the consultation meeting. The methodological issues and challenges, which were ranked in the top 5 by the stakeholders, are (i) outcome measurement, (ii) outcome identification, (iii) cost valuation, (iv) outcome valuation, and (v) time horizon/analytical approach. The existing guidelines and manuals provided guidance for some, but not all, issues and challenges. Discussion and Conclusion: This broad consultation has contributed to a research agenda for the ZonMw programme, which will in the long run lead to the standardization of economic evaluations in this sector in the Netherlands and methodological improvement of economic evaluations in the Dutch youth sector.


2020 ◽  
Vol 6 (4) ◽  
pp. 94
Author(s):  
Pasquale Cacciatore ◽  
Laurenske A. Visser ◽  
Nasuh Buyukkaramikli ◽  
Catharina P. B. van der Ploeg ◽  
M. Elske van den Akker-van Marle

Cost-effectiveness (CEA) and cost–utility analyses (CUA) have become popular types of economic evaluations (EE) used for evidence-based decision-making in healthcare resource allocation. Newborn screening programs (NBS) can have significant clinical benefits for society, and cost-effectiveness analysis may help to select the optimal strategy among different screening programs, including the no-screening option, on different conditions. These economic analyses of NBS, however, are hindered by several methodological challenges. This study explored the methodological quality in recent NBS economic evaluations and analyzed the main challenges and strategies adopted by researchers to deal with them. A scoping review was conducted according to PRISMA methodology to identify CEAs and CUAs of NBS. The methodological quality of the retrieved studies was assessed quantitatively using a specific guideline for the quality assessment of NBS economic evaluations, by calculating a general score for each EE. Challenges in the studies were then explored using thematic analysis as a qualitative synthesis approach. Thirty-five studies met the inclusion criteria. The quantitative analysis showed that the methodological quality of NBS economic evaluations was heterogeneous. Lack of clear description of items related to results, discussion, and discounting were the most frequent flaws. Methodological challenges in performing EEs of neonatal screenings include the adoption of a long time horizon, the use of quality-adjusted life years as health outcome measure, and the assessment of costs beyond the screening interventions. The results of this review can support future economic evaluation research, aiding researchers to develop a methodological guidance to perform EEs aimed at producing solid results to inform decisions for resource allocation in neonatal screening.


2018 ◽  
Vol 38 (7) ◽  
pp. 778-788 ◽  
Author(s):  
Michelle M.A. Kip ◽  
Maarten J. IJzerman ◽  
Martin Henriksson ◽  
Tracy Merlin ◽  
Milton C. Weinstein ◽  
...  

Objectives. General frameworks for conducting and reporting health economic evaluations are available but not specific enough to cover the intricacies of the evaluation of diagnostic tests and biomarkers. Such evaluations are typically complex and model-based because tests primarily affect health outcomes indirectly and real-world data on health outcomes are often lacking. Moreover, not all aspects relevant to the evaluation of a diagnostic test may be known and explicitly considered for inclusion in the evaluation, leading to a loss of transparency and replicability. To address this challenge, this study aims to develop a comprehensive reporting checklist. Methods. This study consisted of 3 main steps: 1) the development of an initial checklist based on a scoping review, 2) review and critical appraisal of the initial checklist by 4 independent experts, and 3) development of a final checklist. Each item from the checklist is illustrated using an example from previous research. Results. The scoping review followed by critical review by the 4 experts resulted in a checklist containing 44 items, which ideally should be considered for inclusion in a model-based health economic evaluation. The extent to which these items were included or discussed in the studies identified in the scoping review varied substantially, with 14 items not being mentioned in ≥47 (75%) of the included studies. Conclusions. The reporting checklist developed in this study may contribute to improved transparency and completeness of model-based health economic evaluations of diagnostic tests and biomarkers. Use of this checklist is therefore encouraged to enhance the interpretation, comparability, and—indirectly—the validity of the results of such evaluations.


2021 ◽  
Vol 21 (1) ◽  
Author(s):  
Yana Seleznova ◽  
Adrienne Alayli ◽  
Stephanie Stock ◽  
Dirk Müller

Abstract Background We aimed to provide a comprehensive overview of methodological challenges in economic evaluations of disease prevention and health promotion (DPHP)-measures. Methods We conducted an overview of reviews searching MEDLINE, EMBASE, NHS Economic Evaluation Database, Database of Promoting Health Effectiveness Reviews, Cochrane Database of Systematic Reviews (CDSR) and Database of Promoting Health Effectiveness Reviews (DOPHER) (from their inception to October 2021). We included both systematic and scoping reviews of economic evaluations in DPHP addressing following methodological aspects: (i) attribution of effects, (ii) outcomes, (iii) inter-sectoral (accruing to non-health sectors of society) costs and consequences and (iv) equity. Data were extracted according to the associated sub-criteria of the four methodological aspects including study design economic evaluation (e.g. model-based), type/scope of the outcomes (e.g. outcomes beyond health), perspective, cost categories related to non-health sectors of society, and consideration of equity (method of inclusion). Two reviewers independently screened all citations, full-text articles, and extracted data. A narrative synthesis without a meta-analysis or other statistical synthesis methods was conducted. Results The reviewing process resulted in ten systematic and one scoping review summarizing 494 health economic evaluations. A lifelong time horizon was adopted in about 23% of DPHP evaluations, while 64% of trial-based evaluations had a time horizon up to 2 years. Preference-based outcomes (36%) and non-health outcomes (8%) were only applied in a minority of studies. Although the inclusion of inter-sectoral costs (i.e. costs accruing to non-health sectors of society) has increased in recent years, these were often neglected (between 6 and 23% depending on the cost category). Consideration to equity was barely given in economic evaluations, and only addressed in six of the eleven reviews. Conclusions Economic evaluations of DPHP measures give only little attention to the specific methodological challenges related to this area. For future economic DPHP evaluations a tool with structured guidance should be developed. This overview of reviews was not registered and a published protocol does not exist.


2020 ◽  
pp. 026921632096759
Author(s):  
Fenella J Gill ◽  
Zahraa Hashem ◽  
Roswitha Stegmann ◽  
Samar M Aoun

Background: Provision of paediatric palliative care is complex and optimally covers meeting the individual needs of a heterogenous population of children and their parent caregivers throughout a life-limiting illness. It is unclear whether existing approaches comprehensively address parent caregivers’ needs. Aim: To examine support needs of parents caring for children with life limiting illnesses and identify specific approaches used to identify and address needs. Design: A scoping review Data sources: MEDLINE, EMBASE, PsycINFO, CINAHL and ProQuest Central, were searched for peer reviewed English language full text research published from 2008 to 2019. Study quality appraisal was undertaken. Fourteen quantitative, 18 qualitative and 12 mixed methods studies were synthesised and themed using summative content analysis and mapped to the Parent Supportive Care Needs Framework (PSCNF). Results: Themes were communication, choice, information, practical, social, psychological, emotional and physical. Communication and choice were central and additional to domains of the PSCNF. Unmet were needs for supporting siblings, for respite care, out of hours, psychological, home and educational support. Six articles reported using instruments to identify parent carer support needs. Conclusion: Support needs of parent caregivers of children with life limiting illnesses are substantial and heterogenous. While studies report evidence of burden and distress in parent caregivers, this rarely translates into improvements in practice through the development of interventions. A systematic and regular assessment of individual parent caregiver support needs is required by using instruments appropriate to use in clinical practice to move the focus to palliative care interventions and improved services for parents.


Politics ◽  
2021 ◽  
pp. 026339572198954
Author(s):  
Yida Zhai

It is widely acknowledged that the economic situation is of vital importance for the stability of an authoritarian regime, but it is rarely known how the public’s economic evaluation contributes to such outcomes. This study examines the effects of citizens’ retrospective and prospective evaluations of their household economic situation and the national economy on the level of regime support in China. The findings show that the national economy outweighs household economic conditions in its effects on the public’s support of the regime. However, the gap between evaluations of the national economy and individual economic situations debilitates regime support. The population in different age cohorts has distinct patterns of relationships between retrospective and prospective economic evaluations and regime support. This study elucidates the political-psychological mechanism of the public’s economic evaluation affecting regime support, and the ruling strategy in authoritarian regimes of manipulating this evaluation.


Author(s):  
Sujita W Narayan ◽  
Ivo Abraham ◽  
Brian L Erstad ◽  
Curtis E Haas ◽  
Arthur Sanders ◽  
...  

Abstract Purpose Cost-avoidance studies are common in pharmacy practice literature. This scoping review summarizes, critiques, and identifies current limitations of the methods that have been used to determine cost avoidance associated with pharmacists’ interventions in acute care settings. Methods An Embase and MEDLINE search was conducted to identify studies that estimated cost avoidance from pharmacist interventions in acute care settings. We included studies with human participants and articles published in English from July 2010 to January 2021, with the intent of summarizing the evidence most relevant to contemporary practice. Results The database search retrieved 129 articles, of which 39 were included. Among these publications, less than half (18 of 39) mentioned whether the researchers assigned a probability for the occurrence of a harmful consequence in the absence of an intervention; thus, a 100% probability of a harmful consequence was assumed. Eleven of the 39 articles identified the specific harm that would occur in the absence of intervention. No clear methods of estimating cost avoidance could be identified for 7 studies. Among all 39 included articles, only 1 attributed both a probability to the potential harm and identified the cost specific to that harm. Conclusion Cost-avoidance studies of pharmacists’ interventions in acute care settings over the last decade have common flaws and provide estimates that are likely to be inflated. There is a need for guidance on consistent methodology for such investigations for reporting of results and to confirm the validity of their economic implications.


2021 ◽  
pp. 152483802098556
Author(s):  
Mark A. Wood ◽  
Stuart Ross ◽  
Diana Johns

In the last decade, an array of smartphone apps have been designed to prevent crime, violence, and abuse. The evidence base of these apps has, however, yet to analyzed systematically. To rectify this, the aims of this review were (1) to establish the extent, range, and nature of research into smartphone apps with a primary crime prevention function; (2) to locate gaps in the primary crime prevention app literature; and (3) to develop a typology of primary crime prevention apps. Employing a scoping review methodology and following Preferred Reporting Items for Systematic Reviews and Meta-Analyses guidelines, studies were identified via Web of Science, EBSCOhost, and Google Scholar. We included English-language research published between 2008 and 2020 that examined smartphone applications designed explicitly for primary crime prevention. Sixty-one publications met our criteria for review, out of an initial sample of 151 identified. Our review identified six types of crime prevention app examined in these publications: self-surveillance apps, decision aid apps, child-tracking apps, educational apps, crime-mapping/alert apps, and crime reporting apps. The findings of our review indicate that most of these forms of primary crime prevention apps have yet to be rigorously evaluated and many are not evidence-based in their design. Consequently, our review indicates that recent enthusiasm over primary crime prevention apps is not supported by an adequate evidence base.


10.5772/56750 ◽  
2013 ◽  
Vol 5 ◽  
pp. 39 ◽  
Author(s):  
Marcello Fera ◽  
Raffaele Iannone ◽  
Vincenzo Mancini ◽  
Massimiliano M. Schiraldi ◽  
Paolo Scotti

The aim of this paper is to present an economic evaluation framework of an RFID system implementation through a pilot project with the aim of streamlining logistic processes and compliance with higher level requirements. The company involved in the project belongs to the CPG (Consumer Packaged Goods) food industry, and in particular to the alimentary industry, whose principal scope was to comply the new norms of the sector by implementation of a better traceability system. This system, besides being very effective for the scope of the project, also had a huge impact in economic terms on logistics resources and cost reduction. For this reason, the discover of the RFID technology for the company has been greatly appreciated at management level, and today other fields of application are under evaluation. The added value of this article is contained not only in the results obtained in the economic evaluations, but also in the applied methodology, evidencing the advantages obtainable in the processes by cutting no added value operations. The economic analysis has thus been conducted on the basis of this TO-BE process streamlining.


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