Search

Genetic relatives of hereditary colorectal cancer patients with Lynch syndrome (LS) are at risk of cancer. Testing both colorectal cancer patients and relatives of mutation carriers for LS allows targeted prevention. However, this could mean disclosing sensitive health data to family members. In light of potential trade-offs between cost-effectiveness and patient privacy, this study investigates the implications of increasing test uptake in Germany. Out of 22 screening strategies for LS, the non-dominated and current German strategies were assessed from the perspective of the statutory health insurance. Life years gained by increased prevention were estimated with Markov models. The effects and implications of different test uptake rates in index patients and their relatives were investigated by scenario analysis. Privacy limitations could yield health gains of up to 2500 undiscounted life years for first-degree relatives of index patients and substantially improve cost-effectiveness. However, this approach may contradict the right to informational self-determination. This study demonstrates the effect higher LS test uptakes could have on the lives and rights of colorectal cancer patients and their relatives. It shows potential conflicts between the efficient use of health care resources on the one hand and reasonable consideration of patient autonomy on the other., (Copyright © 2019 Elsevier B.V. All rights reserved.)

Given the multitude of newly available genetic tests in the face of limited healthcare budgets, the European Society of Human Genetics assessed how genetic services can be prioritized fairly. Using (health) benefit maximizing frameworks for this purpose has been criticized on the grounds that rather than maximization, fairness requires meeting claims (e.g. based on medical need) equitably. This study develops a prioritization score for genetic tests to facilitate equitable allocation based on need-based claims. It includes attributes representing health need associated with hereditary conditions (severity and progression), a genetic service's suitability to alleviate need (evidence of benefit and likelihood of positive result) and costs to meet the needs. A case study for measuring the attributes is provided and a suggestion is made how need-based claims can be quantified in a priority function. Attribute weights can be informed by data from discrete-choice experiments. Further work is needed to measure the attributes across the multitude of genetic tests and to determine appropriate weights. The priority score is most likely to be considered acceptable if developed within a decision process which meets criteria of procedural fairness and if the priority score is interpreted as "strength of recommendation" rather than a fixed cut-off value., (Copyright © 2014. Published by Elsevier Ireland Ltd.)

As our understanding of genetics has increased, so has the number of genetic tests that have entered clinical practice. Given the need of many European health care systems to contain costs, the question of how to prioritise genetic tests fairly has become an emerging concern. This study uses a discrete-choice experiment to assess the value judgements of clinical geneticists, patient representatives and other stakeholders regarding the prioritisation of genetic tests. The respondents chose between two hypothetical scenarios that differed in severity of the disease, risk of the disease, aim of the test, medical benefit of the test, and costs of the test. Standard logit models and mixed effects models were used to estimate the weights different stakeholders attached to attribute levels. Responses from 594 participants were analysed. The most highly valued attribute levels were a proven medical benefit of the test, high risk of having the disease and low costs of the test. Results also showed that rankings differ between clinical geneticists and other stakeholders. The priority weights determined within this study can inform the policy debate and improve the consistency of prioritisation in genetics. Further stakeholder deliberation is needed to explore their most appropriate use in decision practice., (Copyright © 2014 Elsevier Ireland Ltd. All rights reserved.)

The aim of this study was to analyze influences of process- and technology-related characteristics on the outcomes of coverage decisions. Using survey data on 77 decisions from 13 countries, we examined whether outcomes differ by 14 variables that describe components of decision-making processes and the technology. We analyzed the likelihood of committees covering a technology, i.e. positive (including partial coverage) vs. negative coverage decisions. We performed non-parametric univariate tests and binomial logistic regression with a stepwise variable selection procedure. We identified a negative association between a positive decision and whether the technology is a prescribed medicine (p=0.0097). Other significant influences on a positive decision outcome included one disease area (p=0.0311) and whether a technology was judged to be (cost-)effective (p<0.0001). The first estimation of the logistic regression yielded a quasi-complete separation for technologies that were clearly judged (cost-)effective. In uncertain decisions, a higher number of stakeholders involved in voting (odds ratio=2.52; p=0.03) increased the likelihood of a positive outcome. The results suggest that decisions followed the lines of evidence-based decision-making. Despite claims for transparent and participative decision-making, the phase of evidence generation seemed most critical as decision-makers usually adopted the assessment recommendations. We identified little impact of process configurations., (Copyright © 2013 Elsevier Ireland Ltd. All rights reserved.)

Objectives: The comparison of fourth hurdle processes is challenging because they are heterogeneous and decision practice may deviate from formal process rules. This study applies a published framework consisting of key steps of coverage decision processes to the area of cancer prevention., Methods: A research design was developed for analysis of case studies on past decision processes. Decisions were identified and information on the process steps was elicited by semi-structured telephone interviews with decision-makers and experts. The scheme was validated with experts from the areas of screening and prevention and fourth hurdle decision making., Results: Indicators for a structured empirical comparison of coverage decisions were derived. Corresponding ordinal rankings were proposed. Details on six decisions about cancer screening (colorectal and prostate cancer) and vaccination against human papillomavirus in Sweden, Austria and Lithuania are presented., Conclusions: The development of the structured scheme for analysis of coverage decisions allows validation of official statements on decision processes and collection of larger data sets for empirical analysis. However, the semi-structured phone interviews were time-consuming for collecting information on a larger number of decisions. Further validation of the structured scheme and development of a research tool for large-scale empirical studies is still needed., (Copyright © 2011 Elsevier Ireland Ltd. All rights reserved.)

Objectives: It has been claimed that gene technology will induce revolutionary changes in healthcare. This paper investigates how and to what extent these changes have been economically assessed., Methods: A generic framework was developed to distinguish between methodologically similar evaluations of healthcare technology. Methodological issues and the current state of economic evidence concerning human DNA technology were extracted from publications within these groups of evaluations., Results: Economic evaluations of "healthcare consisting of gene technology" were identified primarily for in vitro diagnostics for hereditary disease and others for pharmacogenetics and molecular pathology. "Healthcare enabled by gene technology" is far more encompassing and includes, e.g., biotechnology drugs for which various health economic evaluations can be found. Yet here, the impact of gene technology intertwines with the impact of other technologies and is therefore hardly susceptible to evaluation. The fields of evaluation may be classified best according to the two dimensions "purpose" and "stage of development". Current evaluations cover screening, diagnostic and treatment technologies in investigational, new and established stages. Apart from prenatal screening, healthcare consisting of gene technology was cost saving only for genotype tests replacing continuous phenotype tests and for one pharmacogenetic test. Conclusive evidence of favourable cost-effectiveness ratios is available only for few conditions., Conclusion: Hypotheses about the impact of gene technology on healthcare must be explicit about the definition of "genetic" medicine. A general statement regarding healthcare enabled by gene technology is not possible. Based on current evidence, an era of healthcare consisting of gene technology built on widespread predictive testing is not desirable from a health economic viewpoint.