Background

There has been an increasing awareness of the need to incorporate ethics into the health technology assessment (HTA) process. This need is a consequence of the recognition that addressing moral and ethical issues can increase transparency and accountability of the HTA process and lead to better informed healthcare decisions [1]. As a result, HTA producers and decision-makers are increasingly more interested in considering contextual normative issues and value judgments, in addition to the results of clinical and economic evaluations in HTA.

Despite its importance, integration of ethical aspects into HTA remains challenging for several reasons. One of the key challenges is the plurality of ethical methods that need to be understood by HTA professionals in order to be applied appropriately [2, 3]. Our systematic review of existing guidance documents for ethical analysis in HTA suggested that methods proposed to address ethical issues differ considerably in terms of philosophical approach, structure, and comprehensiveness, and that there is no “one right way” to evaluate ethical considerations around healthcare technologies [4]. Another challenge is that HTA agencies too often fail to adopt the existing ethical guidance documents because most of the guidelines tend towards complexity and call for expertise, time and other resources that might not exist in their organizations. A recent survey of international HTA agencies revealed that only in 15 % of the participating agencies, ethical evaluations were typically performed by individual, mainly external, ethicists or multi-disciplinary teams including ethicists. The majority of the surveyed HTA organization relied on non-ethicist HTA professionals to conduct ethical evaluations, when required [2].

We believe if HTA professionals are expected to take part in ethical evaluations, they need a systematic approach that places greater emphasis on the process and steps required for merging ethics review activities with traditional HTA. This systematic approach should help HTA practitioners better understand the ethical evaluation process, use relevant ethics tools and seek appropriate expert guidance in answering ethical questions, if needed.

A number of existing guidance documents come closest to fulfilling this need. However, they rarely focus on the operationalization of their proposed approaches. For example, the ethical evaluation component of the European network for HTA (EUnetHTA)’s Core model [5], is a comprehensive document that provides a checklist of questions covering ethical issues related to the technology and the HTA process, describes commonly used methods to answer the questions, and proposes a standardized reporting structure. Likewise, the guidelines developed by a number of European HTA agencies, including the Austrian [6], Danish [7], French [8], German [9], Norwegian [10], Spanish [11], and Swedish [12] agencies, promote similar systematic approaches to integrating ethics into the HTA process. However, all of these guidance documents provide few details on what is needed to be done in order to implement the proposed methodology in a routine HTA environment. A further step has more recently been taken by the Swedish Council on HTA to provide a framework that takes into account not only the nature of the HTA process, but also organizational, financial and regulatory elements, as well as the availability of ethical expertise [13]. This framework that focuses on the identification and prioritization of ethical considerations in HTA provides only a brief description for operationalizing the ethics review process.

For HTA researchers with limited experience of performing ethical evaluations, there is still a need for a procedural guidance which would enhance their understanding of how an ethics review can take place during a HTA process and to aid them in incorporating ethical evaluation steps into a typical HTA plan. The aim of this paper is to fill this gap by offering a structured action-oriented framework and a list of literature-driven supporting tools. It should be noted that we do not intend to “reinvent the wheel” by proposing an alternative approach to substitute existing ethical inquiry methods. Rather, our study is intended to provide a process-based framework that encompasses a range of evaluative actions provided by other ethical guidelines for HTA in order to illustrate and describe the steps that are expected to be taken by a HTA team in evaluation of ethical considerations. We believe such a framework would allow HTA producers not only to understand the ethics review process, but also to make use of ethics in their assessments, by identifying interconnections and overlaps between ethics and other domains of HTA. Our framework also brings together the procedural steps and potentially helpful tools to provide more flexibility to the ethical evaluation process in HTA and increase its applicability.

The remainder of this paper is organized as follows: we begin with a description of our multiphase research methodology. Then, we introduce our stepwise framework and explain some important considerations which should be taken into account at each particular step. Next, we introduce a number of the most commonly used tools in ethical evaluation. Finally we offer further discussion of our proposed framework and draw conclusions.

Methods

We initially performed a systematic review of the literature, published up to October 2013, with the purpose of identifying and mapping existing frameworks for ethics in HTA and methodological guidelines from national and international HTA agencies. The review identified 21 methodological articles and 22 HTA guidelines of varying complexity and scope. Data was abstracted, through content analysis, on methodological features of the identified guidance documents, particularly their areas of focus, theoretical foundation, analytical approaches, supporting tools, and required expertise. More details about this phase of research have been published elsewhere [4].

Then, to identify factors that are likely to influence the ethics review process, we conducted a comprehensive search in the literature to create a list of main barriers and facilitators of ethical evaluation in HTA. In addition, we performed a survey of the main HTA producing agencies throughout the world to learn about their experiences, methodological preferences, and their perceptions of the key barriers and enablers to incorporation of ethics in HTA. Ethics approval for the survey was granted by the Hamilton Integrated Research Ethics Board at McMaster University (REB 13-103). Informed consent was obtained using a recruitment email which described the aims of the study and notified participants that completion of the survey implied consent to participate in the survey. Therefore, a separate informed consent form was not required. The findings of the literature review and the survey are published in detail elsewhere [2].

The next stage was to draft a framework based on operational features of the identified guidance documents, as well as practical barriers to avoid and enablers to encourage in performing an ethical evaluation. We used data from the guidance documents included in our systematic review [4] to generate a comprehensive list of main elements and sub-elements that needed to be considered in an ethical evaluation and identify the common elements. The following a priori categories were utilized to group action items: scoping, data collection, analysis, and knowledge translation. However, as the analysis progressed, some preliminary categories were combined and further categories were added. The identified barriers and facilitators were used for further defining and customizing action categories and their related task statements [2]. The process was continued until seven final action categories were established: defining the objectives and scope of the evaluation, identifying stakeholders, assessing organizational capacity, framing ethical evaluation questions, ethical analysis, deliberation, and knowledge exchange or translation.

We generated a separate list of commonly recommended tools, which were identified in our systematic review [4], and examined all in relation to their application and appropriateness for each procedural step. Additional ethical tools were identified through targeted searches in the literature and consultation with subject matter experts. Finally, a summary table consisting of a brief description of the identified tools as well as some of the strengths and weaknesses of each tool was generated.

Results

Our proposed framework consists of an algorithmic flowchart that illustrates a set of steps for operationalizing ethical evaluation throughout various stages of the HTA process; and a stepwise guide, which breaks down each step into individual task objectives and suggests some questions that need to be addressed at each step in order to complete the suggested tasks. To help facilitate the proposed evaluation tasks, a list of some commonly recommended or used tools is also provided, along with a brief description of their strengths and weaknesses. The Framework components are summarized below.

The algorithmic flowchart

As illustrated in Fig. 1, our stepwise flowchart organizes the actions required for an ethical evaluation practice into seven main steps and four conditional steps that allow for revisions and improvements within and across the main steps of the evaluation process. Although in this figure, progression from one step to another is shown to be linear, it is important to note that in real practice ethical evaluation activities can often occur simultaneously or iteratively.

Fig. 1
figure 1

Algorithmic flowchart for ethical evaluation in HTA

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The stepwise guide

To make the framework useful for HTA practitioners, we have attempted to operationalize it into a guide, which can be seen in Table 1. The guide includes a non-exhaustive list of tasks and related questions which are drawn from our systematic review of ethical guidelines for HTA [4], input from experts in the fields of HTA and ethics (n = 6), and from our study of barriers and facilitators of ethical evaluation in HTA [2]. As with many ethics frameworks in other contexts, this guide is intended to aid HTA practitioners in identifying key steps that should not be overlooked, while also helping them to think about possible action items.

Table 1 Stepwise guide for the ethical evaluation process in HTA
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Step1. Defining the objectives and scope of the evaluation

Before starting an ethical assessment, it should be ensured that the objectives of both the overall HTA, to which the ethical evaluation will be incorporated, is set. In doing so, the role of ethics should not be over- or under-estimated. Rather, the scope and aims of ethical evaluation should be proportional to the candidate technology and the context [14]. A careful consideration of potential ethical issues around the primary motivations for conducting an HTA is also required at the scoping step, such as special interests of certain stakeholders in the assessment or external pressure from authorities, manufacturers, and patient groups. Then, as with any other evaluation process, ethical assessment should begin with an exploratory phase to identify the existing knowledge base surrounding the technology of interest such as technological aspects, modes of application, range of possible clinical indications, safety issues, as well as the therapeutic, economic and organizational impacts of the technology. Identifying the purpose of the ethical evaluation is the next essential task, as it will feed into the type of research questions, study design, and choice of ethical analysis approaches.

Step2. Stakeholder analysis

Given the importance of stakeholder interests and values in an ethical evaluation and their influences over potential decisions, it is good practice to conduct a stakeholder analysis during the defining and scoping phase to make sure that values and preferences of potential stakeholders are effectively included in the ethical analysis. Nonetheless, stakeholder analysis can be undertaken throughout all steps of ethical evaluation.

During the scoping phase, potential stakeholders can be identified through brainstorming, collecting and analyzing quantitative or qualitative information, and asking identified stakeholders who they would suggest as relevant stakeholders for the technology of interest. A typical stakeholder analysis involves assessing the interests of the identified key stakeholders, such as patients, healthcare providers, decision makers, family members and the general public in the candidate technology; assessing their importance and level of influence over the HTA process and decision-making; determining who stands to benefit and who stands to lose if the candidate technology is introduced, in what ways and to which extent; and identifying the most appropriate ways to engage stakeholders [15, 16]. Some useful tools for stakeholder analysis are introduced in Table 2.

Table 2 Commonly used tools for ethical evaluation in HTA
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Step3. Assessing organizational capacity

The following resources are necessary in performing an ethical evaluation: a person with a strong educational background and experience in applied ethical theory, sufficient financial resources and time for conducting the evaluation, and the capacity for training, if needed. Therefore, it is important to assess the level of organizational readiness along each of these dimensions in order to balance available resources with the requirements of ethical evaluation.

It must be ensured that sufficient knowledge, experience, and skills exist in the organization to collect ethics-related data and to perform a comprehensive ethical analysis for several reasons. Firstly, since ethical evaluation is an approach which deals with norms and values, conducting such an evaluation would not be possible without a reasonable amount of knowledge of ethical theories and principles. Secondly, because the scope of ethics literature is wide and can include theory as well as both quantitative and qualitative study results, HTA practitioners involved in ethical evaluations should be able to effectively appraise ethics literature and reflect on the collected information. Thirdly, a well-performed ethical analysis uses moral reasoning rather than merely describing facts and values. Hence, the rigour of ethical reasoning is usually dependent on how skillfully the analysis has been performed. Furthermore, the capacity in methodologies associated with ethical analysis through public discourse is often lacking in some HTA organizations [17]. Therefore, to get involved in public participatory processes, HTA professionals might need to acquire a range of new skills in different methods of public engagement before getting involved in such research activities [14, 18].

Step4. Framing ethical evaluation questions

Recognition of existing ethical dilemmas or the ones that are perceived likely to emerge after implementation of the technology (hypothetical dilemmas) is essential for the formulation of ethical questions that need to be answered. Identification of the existing ethical issues that could be resolved through the introduction of the technology should also be accounted for. Zydziunate et al. [19] systematically reviewed ethical dilemmas that might affect decision-making within health care systems and suggested that ethical dilemmas in healthcare might happen in institutional, local or national levels. The review listed the following terms that had been commonly used in defining or discussing ethical dilemmas: “continuing balancing” between health care needs and budgets, “result of resource allocation”, “gap between professional obligations and possibilities”, “ethically controversial situation”, “concern about interactions”, “outcome of medical choices”, “concern about society’s access to healthcare resources”, and “ethically difficult or ethically challenging situation”.

In practice, it might not always be necessary to make a comprehensive list of existing ethical conflicts or controversial issues through a systematic inquiry. However, it is important to discuss and specify which of the recognized ethical considerations and arguments are more relevant to the assessment and provide a justification for why these could be relevant. One familiar example is the dilemma that might arise in situations involving genetic testing technologies. Genetic test results, by nature, can reveal aspects of the tested individual’s susceptibility to health problems (potential for stigmatization or discrimination). In addition, they may have implications on the blood relatives of the tested individual, who might also request to know their family member’s genetic test results (potential for information abuse or intrusion of privacy). These aspects of the technology should be explored and discussed while framing the ethical evaluation questions. At this phase of evaluation, a priority should be assigned to ethical questions that may have greater implications on decision making. However, the HTA team must allow flexibility for adding questions during the ethical evaluation process.

Step5. Ethical analysis

As it was previously mentioned, the present framework does not aim to provide instructions on how to perform an ethical analysis, but rather assumes that HTA team members who are responsible for the analysis of ethics data have the knowledge and skills to take on this important task. Ethics expertise can be critical at this step, depending on the type of the assessment. A normative (principle- or theory-based) evaluation should generally be performed with the help of experts with knowledge in ethical theory. In participatory or interactive assessments, where expert and lay opinions are considered equally valuable, ethicists can play an active role by providing rationale for potentially useful analytical approaches, scientific and theoretical inputs to stakeholder and public debates and assisting stakeholders in reaching a consensus [14]. In sensitive topics, it may be desirable to seek discussion from more than one ethicist. Other HTA practitioners (non-ethicists) can also have a role, although necessarily limited, in ethics review and analysis (e.g., helping with formulating ethical questions and searching for potential solution through systematically identifying and summarizing ethics-related data, helping with participatory research, etc.).

Our systematic review identified multiple guidance documents for the analysis of ethical data. The identified methodological approaches vary in their goal, philosophical approach, structure, and comprehensiveness. Traditional approaches consist of making ethical issues explicit by identifying facts and values, setting out arguments, providing reasons and justifying potential decisions through moral principles and theories (e.g., principlism, deontology, casuistry, and axiology). Deliberative approaches, on the other hand, advocate participatory and interactive approaches as complementary methods to traditional normative reasoning (e.g., wide reflective equilibrium, actor-network theory, interactive HTA, and social shaping of technology). It can be useful to employ more than one method in a given evaluation to help attend to the problem of bias, address the complexity of ethical dilemmas and uncertainties around healthcare technologies, and better justify HTA decisions [14, 20–23]. Different approaches can be compared and contrasted to more thorough and balanced results.

Hofmann et al. describe the most frequently used approaches for addressing ethics in HTA and provide guidance for choosing among various analytical approaches [24]. They believe that the selection of analytical approach for addressing ethical considerations in a specific HTA depends on the goal, context and process of HTA. Based on their classification of different methods of integrating ethics into HTA, the authors of this guidance document suggest that traditional bioethical approaches (e.g., deontology, casuistry, principlism, and axiology) are more suitable if ethical inquiry is performed as an independent assessment with less (subsidiary integration) or equal (combined integration) importance compared to other domains of HTA; while the approaches that are developed with a focus on the process of HTA (e.g., Socratic approach, interactive technology assessment, and social shaping of technology) are more appropriate when there are overlapping dynamics (coordinated integration) or continuous interactions (interactive integration) between ethics and other domains of HTA.

It is important to note that the results of ethical analyses may be influenced by the analysts’ knowledge, their experiences, values, and attitudes, as well as the technological, organizational, social, and political contexts in which the analyses are performed. Ethical analyses are also at risk of bias due to actual or potential conflict of interests in the HTA organization, such as which may influence the ways in which ethical information is collected, analyzed and interpreted. Therefore, it is good practice to provide a sound justification of the choice of the methods and disclose any financial and non-financial relationships with organizations or groups who may have an interest in the candidate technology and its implementation.

Step6. Deliberation

Once the preliminary analysis of ethical data is completed, it is desirable to discuss the results with the members of the multidisciplinary HTA team, and other experts if needed, in order to verify plausibility and reasonableness of the results. To ensure that the results are perceived as relevant by stakeholders and the public, it is also important to use public engagement methods to take in a variety of inputs from the groups whose values and preferences can provide a means for a better informed and legitimate policy decisions [25]. Although engagement of the public (including relevant stakeholders) in an ethical evaluation process, and in HTA in general, has been promoted at different levels, the common exercise is to gather the public input in an ad-hoc basis through deliberative methods such as surveys, focus group discussions, etc. Alternatively, the public input can be sought directly from the public representatives who are involved throughout the HTA process, or through an institutionalized approach, where the public or specific stakeholder groups are asked, as consultants, to provide input for decision-making in an ongoing basis [25]. Despite its importance, as a democratic exercise in ethical evaluation, public involvement methods can be challenging to perform due to their complexity, costliness, and time consuming nature [26].

Additional expert insight might be necessary to ensure the plausibility of the produced results during stakeholder hearings, before a particular conclusion is reached and the final report on ethical issues around the technology is written.

Step7. Knowledge exchange/translation

The purpose of HTA is primarily to support healthcare policy-makers in making evidence-informed decisions, and secondarily to help advance knowledge about a particular health technology and stimulate further research [27]. Therefore, the dissemination of the HTA findings, including ethical aspects, must be timely and appropriately tailored to the needs of potential users.

In order for the results of an ethical assessment to be utilized as an input for decision-making, the knowledge translation activities should begin in the earliest stages of the HTA process, through an effective interaction between HTA-producers and decision-makers, and continue throughout the evaluation [28]. The results need to be communicated in a manner that can be understood and easily utilized by decision-makers. The feedback from potential users should be received throughout the project and used to improve the quality of the research. HTA reports should address various dimensions of an existing or hypothetical ethical problem surrounding the technology of interest, using all relevant evidence from research and non-research sources, and applying suitable analytical approaches. It is also important to address how different stakeholders and members of society might be affected by the implementation of the technology or otherwise.

HTA findings can also be disseminated among other relevant target groups, such as healthcare researchers, clinicians, healthcare service providers (e.g. hospitals), third party payers, biomedical manufacturers, patients, and the general public. However, it is essential to translate the findings (including the results of the ethical analysis) into formats that are understandable and useful to the above-mentioned groups of audience [7].

Selected tools

Table 2 provides a list of tools and techniques to support ethical evaluation tasks. This list does not include every possible tool that could potentially be used at each step of the proposed model, but has explored some of the more common ones that can assist HTA practitioners in evaluation of ethical considerations. It is important that HTA practitioners with responsibility for addressing ethical issues have a comprehensive knowledge and understanding of such tools in order to choose tools which are appropriate for varied types of ethical objectives and evaluation tasks.

Four categories of tools can be distinguished in Table 2: ethics literature review and appraisal, stakeholder analysis, exploring stakeholder values and preferences, and identification and analysis of ethical issues. We also searched for computerized support tools for aiding ethical analysis and identified a number of tools which were designed to help the users in summarizing and structuring ethical arguments, describing potential inter-relations between the interests of different stakeholders, or analyzing the impact of alternative technologies on various stakeholders’ interests. Although the selected computer programs, i.e., EthXpert [29] and Ethos [30], were originally offered as ethical decision-making support tools, they could potentially be applicable in ethical analysis for HTA.

Discussion

We believe that, only by fully understanding all of the different steps of ethical evaluation and specific issues that may arise at each step, can HTA teams integrate ethics in their assessments. The framework presented in this article outlines the important steps that should be adopted by HTA practitioners for a comprehensive evaluation of ethical considerations. It is intended to guide HTA producers, especially those who are not accustomed to performing ethics reviews. The framework may also be used by researchers, evaluators, or decision-makers in order to critically appraise the process used for an ethical evaluation; or for educational purposes, especially to show the flow of activities needed for the evaluation of ethical considerations. Our framework may be a beneficial addition to the existing literature on ethics and HTA, for the following reasons:

Firstly, the framework is informed by the key operational features of existing ethical guidelines for HTA, as well as practical concerns and technical demands of potential user. It recognizes that ethical evaluations might be discounted or not undertaken in some HTA organizations because they are perceived as being impractical, resource consuming, or unfeasible. Therefore, the framework encourages proper scoping, strategic resource planning, and strengthening organizational capacity.

Secondly, the proposed framework aims to promote a more systematic and structured way of integrating ethics into HTA by mapping the relatively complex process of ethical evaluation and highlighting its main steps. Nonetheless, it is conceptually simple and employs terms and concepts that are familiar to the majority of HTA practitioners, including those who are less familiar with ethical evaluations. The visual representation of the stepwise process helps HTA practitioners to understand the nature of an ethical evaluation. The stepwise guide, on the other hand, reduces the complexity of evaluation by breaking down the procedural steps to smaller sets of tasks and providing guiding questions. With its focus on making the evaluation process more understandable and practical for all HTA practitioners, the framework can be easily used in training of HTA professionals, as well as also being able to play a role in harmonizing existing evaluation approaches across HTA organizations.

Thirdly, the stepwise framework accommodates the changing relationship between technology assessment, policy, and society by fostering the integration of stakeholder and public input into the ethical evaluation process. Furthermore, it promotes a holistic approach to evaluation and stresses that ethical issues need to be seen in interconnection with clinical, economic, social and legal issues, and that these relationships require an appropriate cooperation between the team members working in different domains of HTA. Another important aspect of the stepwise framework is its flexibility. The users of the framework are offered the possibility to choose the tasks that are more relevant to their assessments or to customize the evaluation process according to their needs.

In summary, the strengths of our proposed framework lie in its structured yet flexible approach to the evaluation of ethical considerations in HTA. It can, therefore, be helpful in building a more consistent practice of ethical evaluation among HTA professionals.

In spite of its strengths, our proposed framework also has limitations. Firstly, to use the framework HTA producers may require information, data and expertise and other resources that may not be readily available in a typical HTA organization. Qualitative evidence, stakeholder input and normative judgements are usually required to address ethics-related questions. Our framework fails to deeply address the ways in which ethical data should be tackled or specify how the evaluation process should be applied in different contexts. Secondly, the current version of our stepwise framework lacks an example case study where all the steps are applied. Furthermore, the validation of the proposed framework was not performed as part of this research project.

We will undertake further work to validate the framework and test its practicality through case studies, seeking stakeholder feedback, and expert opinion. Based on the results from the case studies and feedback received from the experts and potential users, we are planning to: (a) modify the stepwise framework by adding or removing steps; and (b) enhance its practicality by adding flow charts that illustrate details of various steps, and auxiliary tools or checklists to facilitate the ethical evaluation process; and (c) perform a final validation of the framework using further case studies.

Conclusions

Despite the increasing attention given to the incorporation of ethical considerations in assessment of healthcare technologies, HTA producers continue to face challenges in integrating ethics to HTA. The intention of this research has been to construct a procedural framework that considers the nature and sequence of ethical evaluation process in the context of HTA. The proposed framework provides a conceptual foundation to allow for ethical issues to be addressed in HTA. Our framework can serve as a starting point towards a set of comprehensive strategic guidelines, as well as for the supporting and promoting good practice of integrating ethics in HTA. However, for a wider use and dissemination, its content needs to be applied in various HTA projects and validated through consultation with experts and policy makers.

Abbreviations

EUnetHTA, European Network for Health Technology Assessment; HTA, Health Technology Assessment