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Purpose: Exome or genome sequencing (ES or GS) can identify genetic causes of otherwise unexplained congenital anomaly and perinatal death (PND) but is not routine practice. The evidence base for "genomic autopsy" after termination of pregnancy for fetal anomaly (TOPFA) and PND has been synthesized to determine the value of this investigation., Methods: We conducted a systematic review and meta-analysis of studies meeting prespecified inclusion criteria and containing ≥10 cases of TOPFA or PND (with or without major congenital abnormality), in which ES or GS was conducted. We determined test performance, including diagnostic yield, accuracy, and reliability. We also reported outcomes associated with clinical utility and harms, where described., Results: From 2245 potentially eligible studies, 32 publications were eligible and had data extracted, representing 2120 cases that could be meta-analyzed. No diagnostic accuracy or comparative studies were identified, although some analysis of concordance between different ES/GS methodologies could be performed. Studies reporting parent-related outcomes or long-term follow-up did not do so in a systematic or quantifiable manner., Conclusion: Evidence suggests that approximately one-fourth to one-third of fetal losses associated with TOPFA or unexplained PND are associated with a genetic cause identifiable on ES or GS-albeit this estimate varies depending on phenotypic and background risk factors. Despite the large body of evidence on ES and GS, little research has attempted to validate the accuracy of testing, nor measure the clinical or societal outcomes in families that follow the diagnostic investigation in this context., Competing Interests: Conflict of Interest Camille Schubert’s salary was partially funded by the Genomic Autopsy Study (Australian government MRFF grant funded research). All other authors declare no conflicts of interest., (Copyright © 2024 The Authors. Published by Elsevier Inc. All rights reserved.)

Objectives: Computerized clinical decision support software (CDSS) are digital health technologies that have been traditionally categorized as medical devices. However, the evaluation frameworks for traditional medical devices are not well adapted to assess the value and safety of CDSS. In this study, we identified a range of challenges associated with CDSS evaluation as a medical device and investigated whether and how CDSS are evaluated in Australia., Methods: Using a qualitative approach, we interviewed 11 professionals involved in the implementation and evaluation of digital health technologies at national and regional levels. Data were thematically analyzed using both data-driven (inductive) and theory-based (deductive) approaches., Results: Our results suggest that current CDSS evaluations have an overly narrow perspective on the risks and benefits of CDSS due to an inability to capture the impact of the technology on the sociotechnical environment. By adopting a static view of the CDSS, these evaluation frameworks are unable to discern how rapidly evolving technologies and a dynamic clinical environment can impact CDSS performance. After software upgrades, CDSS can transition from providing information to specifying diagnoses and treatments. Therefore, it is not clear how CDSS can be monitored continuously when changes in the software can directly affect patient safety., Conclusion: Our findings emphasize the importance of taking a living health technology assessment approach to the evaluation of digital health technologies that evolve rapidly. There is a role for observational (real-world) evidence to understand the impact of changes to the technology and the sociotechnical environment on CDSS performance.

There is currently deep uncertainty about the clinical benefits and cost effectiveness of highly specialised technologies (HSTs), like gene and cell therapies. These treatments are novel, typically have high upfront costs, the patient populations are small and heterogenous, there is minimal information on their long-term safety and effectiveness, and data are limited and often of poor quality. With the increasing number of these technologies and their high cost burden on governments and health care providers, policy makers are currently walking a decision tightrope. On the one hand, an unfavourable funding decision could potentially limit patient access to life-saving treatments, while on the other, a favourable decision could result in unsustainable budget impacts and perhaps poorer patient health outcomes. Health technology assessment (HTA) is meant to determine the value of a health technology in order to promote an equitable, efficient, and high-quality health system. However, standard HTA processes have failed to mitigate the deep uncertainties associated with these technologies. In this paper, we propose a Living HTA framework to address these challenges. This framework includes a one-off process for making explicit the societal values associated with HSTs. These would inform the decision-making approach, data collection and the development of disease-specific reference models to be used by industry sponsors as the basis for their submissions for public funding. Coverage with an evidence development mechanism is also proposed by which data can be collected in real time to update the reference model on a rolling basis, thereby allowing re-assessment of the clinical and cost effectiveness of individual HSTs. The HTA would be 'live' until the results indicate there is sufficient certainty for the funding decision to be confirmed, the price changed or the funding removed., (© 2023. The Author(s).)

Background: Although the importance of clinical ethics in contemporary clinical environments is established, development of formal clinical ethics services in the Australia health system has, to date, been ad hoc. This study was designed to systematically follow and reflect upon the first 18 months of activity by a newly established service, to examine key barriers and facilitators to establishing a new service in an Australian hospital setting., Methods: HOW THE STUDY WAS PERFORMED AND STATISTICAL TESTS USED: A qualitative case study approach was utilised. The study gathered and analysed data using observations of service committee meetings, document analysis of agendas and minutes, and semi-structured interviews with committee members to generate semantic themes. By interpreting the thematic findings in reference to national capacity building resources, this study also aimed to provide practice-based reflections for other health agencies., Results: THE MAIN FINDINGS: An overarching theme identified in the data was a strong commitment to supporting clinicians facing difficult patient care decisions and navigating difficult discussions with patients and families. Another key theme was the role of the new clinical ethics support service in providing clinicians with a pathway to raise system-wide issues with the organisation Executive. While there was strong clinical engagement, consumer and community participation remained a challenge, as did unresolved governance issues and a need for clearer policy relationship between the service and the organisation. Considering these themes in relation to the national capacity building resources, the study identifies three areas likely to require ongoing development and negotiation. These are: the role of the clinical ethics support service as a link between the workforce and the Executive; the incorporation of consumers and patients; and ethical reasoning. To improve the effectiveness of the service, it is necessary to increase clarity on the service's role at the governance and policy level, as well as develop strategies for engaging consumers, patients and families. Finally, the capacity of the service to reflect on complex cases may be enhanced through explicit discussions of various different ethical frameworks and ways of deliberating., (© 2023. BioMed Central Ltd., part of Springer Nature.)

Due to the increasing threat to public health and the economy, governments internationally are interested in models to estimate the future clinical and economic burden of antimicrobial resistance (AMR) and to evaluate the cost-effectiveness of interventions to prevent or control resistance and to inform resource-allocation decision making. A widely cited UK report estimated that 10 million additional deaths will occur globally per annum due to AMR by 2050; however, the utility and accuracy of this prediction has been challenged. The precision of models predicting the future economic burden of AMR is dependent upon the accuracy of predicting future resistance rates. This paper reviews the feasibility and value of modelling to inform policy and resource allocation to manage and curb AMR. Here we describe methods used to estimate future resistance in published burden-of-disease models; the sources of uncertainty are highlighted, which could potentially mislead policy decision-making. While broad assumptions can be made regarding some predictable factors contributing to future resistance rates, the unexpected emergence, establishment and spread of new resistance genes introduces substantial uncertainty into estimates of future economic burden, and in models evaluating the effectiveness of interventions or policies to address AMR. Existing reporting standards for best practice in modelling should be adapted to guide the reporting of AMR economic models, to ensure model transparency and validation for interpretation by policymakers., (© 2022. The Author(s).)

Objective: Governments around the world face challenges in maintaining sustainable, high-quality healthcare systems. Health Technology Assessment (HTA) is widely used as a method to assist in funding decisions. However, the scope and influence of HTA is still limited. We examined how policymakers can improve the usefulness of HTA., Methods: We used Systems Thinking as a theoretical framework to examine HTA as a system. We purposely sampled stakeholders involved in Canadian HTA at a national or provincial level. We conducted 22 semi-structured interviews in September-December 2016. Data were analysed using NVivo10 and findings are presented as a concept map with explanatory text., Findings: The HTA system is influenced by stakeholder interactions. Such interactions are, in turn, affected by stakeholders' worldviews and environmental factors. Stakeholders' worldviews includes individual's or groups' values and affect the exchange of information, and interpretation of events. External factors, such as changes to government structures, also affect the system., Conclusion: Most stakeholder groups are supportive of the system. However, participants identified a need for change, though the exact changes being recommended differed. Some interactions were praised (formal, inclusive collaborations to provide government with policy guidance on both broad and technology-specific matters), while other interactions were criticised (two-party alliances formed around purposes other than the common good, and lacklustre patient and industry engagement on the part of provincial government)., Competing Interests: Declaration of Competing Interest This project was undertaken as part of EL's PhD project. EL received funding from the Australian Department of Education and Training (Endeavour Fellowship) and had some travel costs covered by The University of Alberta (Canada). JS, DC, and TM declare that they do not have a conflict of interest with this project. TS has participated in Health Technology Assessment for the pan-Canadian and provincial processes but declares that she does not have a conflict of interest with this project., (Copyright © 2020 Elsevier B.V. All rights reserved.)

Objectives: A mapping exercise was undertaken to determine how HTA is being described and conducted across the International Network of Agencies for Health Technology Assessment (INAHTA), with the aim of harmonizing terminologies and approaches., Methods: Three progressive surveys were undertaken. In 2010, INAHTA agencies were asked to provide details on all of their HTA products. In 2013, additional information was sought on key methodological characteristics of five of the most common HTA product types. Subsequently, final agreement was sought on three proposed product types., Results: Forty-five HTA agencies responded to at least one of the surveys. In 2010, twenty-one agencies reported publishing over seventy named HTA products. Core domains associated with full HTA reports were reported by a third of agencies but were labeled differently, so products were classified according to product type (n = 17). Agencies producing short, tailored products increased between 2010 and 2013, with the publication of rapid reviews doubling from 33 percent to 66 percent. In 2013, half of the agencies adapted their common HTA products from documents produced by other agencies. A consensus (>70 percent) was achieved on definitions for HTA reports, mini-HTAs, and rapid reviews., Conclusions: The product label for an HTA is not always indicative of its content. Terminology has, therefore, been agreed to make explicit the trade-off between rigor and timeliness in three common HTA product types. An INAHTA Product Type (IPT) Mark has been created to identify each of these. It is hoped this will further facilitate HTA adaptation between agencies and reduce duplication of effort.