Introduction
The prevalence and requests for medical treatment of gender dysphoria (GD) have risen in many countries in the last decade. In Finland, for example, 2–4 percent of adolescents reported possible clinically significant GD in 2017; treatment seeking was much more rare, but increased fivefold in between 2011 and 2017 (Reference Kaltiala-Heino, Työläjärvi and Lindberg1). Discussions about medical treatments of GD have created large societal interest also outside health care and health policy. Creating widely acceptable treatment guidelines on the topic is important but complex, for several reasons.
First, GD can be highly stigmatizing and lead to discrimination (Reference Kcomt2;Reference Bradford, Reisner, Honnold and Xavier3). Attitudes of healthcare professionals and availability of medical treatments for GD can be important mediators of stigma. Creating treatment guidelines that are both stigma-reducing and evidence-based is important.
Second, the categorization of the phenomena is changing. Professional terminology and diagnostic labels are important mediators of both availability of health services and social stigma. WHO’s ICD-10 classification includes a category of gender identity disorders (GID) under the class of mental disorders. This includes transsexualism (adults or adolescents) defined as “a desire to live and be accepted as a member of the opposite sex, usually accompanied by a sense of discomfort with, or inappropriateness of, one’s anatomical sex, and a wish to have surgery and hormonal treatments to make one’s body as congruent as possible with one’s preferred sex” (4). In contrast, the more recent American Psychiatric Associations DSM-5 classification uses the term GD (5) and ICD-11 uses gender incongruence (6), but neither includes a category of transsexualism. ICD-11 moves gender incongruence from mental disorders to a new category called conditions related to sexual health. This paper and the health technology assessment (HTA) it is based on uses the term GD.
Third, increase in requests for treatment has created challenges in meeting the demand and an interest in clarifying diagnostic and treatment protocols (7).
Fourth, GD is approached quite differently in different countries. Although cultural and legal issues should not influence summarizing empirical evidence, they inevitably influence how people with GD are met on societal level and how national treatment guidelines are created. The importance of culturally sensitive and value-based interpretations of evidence is emphasized when empirical evidence is far from conclusive.
Thus, creating transparent, justifiable, and widely acceptable treatment guidelines for GD is both highly important and unusually complex. As a guideline on a topic like this cannot rely purely on value-neutral review of empirical evidence, identification of ethical issues, and transparent balancing of different stakeholders’ values is called for (Reference Saarni, Hofmann and Lampe8).
The Finnish health care is primarily tax-funded and organized by municipalities. The Finnish Council for Choices in Health Care (COHERE) is attached to the Ministry of Social Affairs and Health, and tasked to give recommendations about services included in publicly funded health care. Between 2018 and 2020 COHERE conducted a comprehensive HTA of medical treatments of GD with an integrated ethical analysis, the first we are aware of, and issued a recommendation on the topic.
This paper presents the process of integrating ethical analysis into the HTA, and the consequences this had for the process and the results. Term “HTA” is used in this paper to include both technology assessment and appraisal phases.
All documents, including English and Swedish summaries and the forty-page ethical analysis report (in Finnish), are found on COHERE website (www.palveluvalikoima.fi/en/recommendations). A detailed English summary of the ethical analysis has been submitted.
Methods
The EUnetHTA Method of Integrated Ethical Analysis
The EUnetHTA Core model (9) includes not only effectiveness, safety, and costs, but also ethical, organizational, social, and legal aspects as core parts of HTA (Reference Lampe, Mäkelä and Garrido10). The ethical analysis consists of three tasks: (i) exploring and mapping out all the relevant ethical issues, (ii) analyzing these issues with different ethical theories, and (iii) integrating the value-analyses within the whole HTA process.
The EUnetHTA model includes a list of topics (“cards”) that are used to identify, analyze and present the relevant ethical issues in a structured and transparent way. This facilitates transfer of information and transparent use of ethical considerations in evidence appraisal. The ethics model includes six general topics: balance between benefits and harms, autonomy, respect for persons, justice and equity, legislation, and the ethical consequences of the HTA.
Integrated ethical analysis contrasts with ethics as an “add-on” or with having no explicit ethical analysis. An add-on model in practice means that professional ethicists compile an independent report assessing ethical issues. This report is considered at the latest by those appraising the evidence and creating the recommendation. Many value decisions must be made during the HTA process. Not having explicit ethical analysis does not mean that ethical issues are not considered, but usually that they are not systematically considered or documented.
Integrating ethics means that ethical experts are members of the HTA team and ethical issues are considered during the entire process. The whole HTA team participates in discussing ethics. The definition of ethical expertise is not straightforward but both experts (S.I.S. and S.U.) fulfill the criteria recently presented (Reference Refolo, Bond and Bloemen11).
Planning the HTA and the Ethical Analysis
Ethical expertise was considered useful due to the complex and controversial nature of the topic. GD fulfills all five characteristics of complexity that emphasize the importance of ethical analysis, as proposed by the INTEGRATE-HTA project: multiple important perspectives, difficulty of defining the phenomena, uncertain causality, unpredictable outcomes, and ethical complexity (Reference Lysdahl and Hofmann12, Reference Lysdahl, Mozygemba and Burns13).
A separate legal or social analysis was omitted because of the ongoing legal reform, but the person responsible for that joined the workgroup. The legislation in Finland is from year 2002, and only covers how an adult can change his/her legal gender.
A workgroup with twenty-two persons was formed by inviting experts from the two University hospitals treating GD in Finland. Resulting multidisciplinary workgroup included psychologists, a pediatrician, adolescent and adult psychiatrists, gynecologists, and plastic surgeons, all with long clinical and/or research history in the field, plus lawyers, HTA experts, and ethicist. Patient and other stakeholder hearings were planned from the start, but COHERE protocol does not include patients as official members of workgroups. Although written by the COHERE staff, the workgroup as a whole is responsible for producing the HTA, which is finally approved by COHERE board. The ethical analysis reports, like literature review and separate health economics analyses, are supporting documents. Although these are also accepted by the board, they are not part of the official guidance.
Analyzing the Contribution of Ethics in HTA
Although some evidence suggests that different ethical methods can lead to relevantly similar conclusions (14), it is important to understand how integrating or omitting ethical analysis influences the HTA. An inherent consequence of the integrated model is, however, that the exact contribution of ethical expertise cannot be unambiguously defined.
EUnetHTA model encourages ethical discussions in all the three phases of the HTA:
Planning:
– Considering ethical issues related to why the HTA is conducted, potential interests of different stakeholders and participants.
– Scoping the HTA, defining the endpoints and research methods in light of morally relevant issues.
Evidence gathering:
– Gathering data for the ethical analysis report from literature, content experts, and stakeholders. Participating in data gathering of other workstreams, like effectiveness.
Summary and appraisal:
– Balancing the ethical topics identified for the ethics report.
– Including ethical considerations in the appraisal of empirical evidence and recommendation formulation.
– Public commentary.
This paper describes the ethical discussions and their consequences following the above structure.
Results
Planning Phase
Considering the Ethical Issues Relevant for Conducting the HTA
Many ethically relevant issues supporting a timely HTA were identified in planning phase – discussions with the ethicists. The treatment of GD was centralized into two university hospitals, but both struggled to meet the demand and maintain equal treatment policies, based on common understanding of scientific evidence. The law governing transgender rights was under review, and NGOs were demanding a more liberal treatment policy. Identifying the various ethical themes confirmed the importance of including a formal ethical analysis in the process.
Scoping and Defining Research Methods
HTA typically employs a “PICO” approach, meaning that the patients, intervention, control group, and outcomes should be clearly and unambiguously defined before literature search. Choosing these constitutes a value decision that defines the whole HTA, as it limits what kind or literature is identified, and what outcomes are considered valuable.
Scoping discussions suggested that the outcomes patients hoped to achieve with medical treatments seemed to be complex and multidimensional. Important outcomes could be biological and functional (e.g., body shape, structure, and function), psychological (e.g., decreased dysphoria), and social (e.g., living in a new gender role accepted by the society). Summarizing such multidimensional outcomes was presumed difficult. This discussion led to keeping the literature search as wide as possible, that is, not excluding studies by strict quality criteria or lack of predetermined outcome measures.
Terminology and diagnostic classification were repeatedly discussed during the HTA. Diagnostic labels with regards to GD carry significant symbolic value, but terminology also has social, policy, and health coverage implications. Research on the topic is mostly based on ICD-10 and DSM classifications, but future health care is likely based on ICD-11. All use different terminology and categorizations. Although medical treatments in Finland are officially indicated for ICD-10 GIDs, the HTA was originally titled medical treatments of dysphoria related to gender variations, to find a widely acceptable terminology that would be future-proof and cover the issues where HTA-based guidance was needed. During the process, however, the recommendation was divided into three and labeled: recommendations on medical treatment methods for GD related to “transgender identity in adults” (ICD-10 F64.0) “nonbinary gender identity in adults” and “variations in gender identity of minors.” This structure was considered pragmatic from the health-system point of view, because treatment paths, empirical evidence base, and legal status for these three groups differed somewhat. Legal sex change is possible only for adults and surgery is not available for minors.
Evidence Gathering Phase
The EUnetHTA model gathers ethics data by analyzing the content and relevance of 19 ethical issues sorted under six topics.
The literature search and summary on empirical topics were conducted by a company specializing in health informatics and HTA (Summaryx Ltd). The literature search strategy is presented in the Supplementary Material. General searches were conducted on 20 December 2018 on Pubmed, CLIB, Scopus, and Psycinfo and complemented regarding psychosocial interventions on 20 February 2019. Search results list was received by the ethicists, and the company was asked to pick out articles containing ethical themes. The searches yielded 2,088 hits, with 232 articles retrieved for analysis. Although eleven terms were used to identify GD-related papers in all searches, an additional ninety-three papers were identified outside the searches, reflecting the complex terminology. In total, 325 articles were included in the literature review. In addition, separate literature searches were conducted on PhilPapers and philosophy of medicine and bioethics journals by S.U. to identify nonmedical papers.
The ethicists drafted key points for each ethical topic based on the literature. Drafts were presented to the workgroup for discussion on several meetings. Discussions aimed to identify issues that were the most relevant, potentially controversial, or those where the ethicists needed a deeper understanding of the empirical issues.
Themes brought up for discussions were:
1. How to identify what symptoms and outcomes are important for the patients, clinicians, and evidence appraisal. How could biological, psychological, and social outcomes be balanced or summarized?
2. What are the morally relevant differences between dysphoria of people with trans- and cis-gender identities? Body-modifying treatments of persons with cis-gender identity are not usually publicly funded in Finland. Question of equality, justice, and resource allocation were discussed.
3. Discussing GD brings up complex emotions, prejudices and might challenge traditional professional roles. The tensions between seeing gender identity as a medical, social, or human rights issue were discussed. HTA was proposed as a practical way to reconcile these different discourses, or at least clarify the roles and responsibilities of different stakeholders. This idea was accepted but turned out difficult to achieve in practice.
Themes brought up by clinicians that influenced the ethical analysis were:
4. Quality of empirical evidence was low, especially considering the clinical practice. The possibility for serious bias in follow-up studies using subjective satisfaction as an outcome without a control group, that were often presented as evidence of effectiveness.
5. Clinicians identified a distinct group of adult transgender men and women, the treatment of whom was considered as fairly uncontroversial (Reference Gerritse, Hartman and Antonides15). Thus, the HTA was especially needed to clarify the treatment of minors and nonbinary adults.
6. The importance of psychosocial support for children and adolescents. Treatment seekers include a group whose dysphoria may not be primarily about gender, or who do not wish to continue to medical treatments after their dysphoria has alleviated. The goal should be to support the autonomy of adolescents, but autonomous decision making is not easy, as dysphoric adolescents must make complex and life-defining choices. Competence and informed decision making can and should be actively supported.
7. Professionals consider giving treatments with permanent effects for people who would later regret them as very serious mistakes. This is put under the maxim “primum non nocere”; biomedical treatments are conceptualized as altering a healthy body, and thus as harmful until evidence shows otherwise. This exemplifies an important value conflict: requiring high-quality evidence of benefits can contrast with the view that prioritizes autonomy and informed risk-taking.
8. The need for cooperation in the multidisciplinary decision-making process between different medical specialists. For example, the decision making of psychiatrists diagnosing, and plastic surgeons operating have different grounds (diagnostic criteria, risks of surgery).
9. The political nature of the discussion about gender identity and minority rights. Current evidence, healthcare legislation, and strong emphasis on patient rights can be interpreted differently with different priorities.
These value-themed discussions gave important insights to the ethicists but were also an important facilitator of consensus within the workgroup. Despite this, differences in opinion also existed within the group, both between physicians of different specialties, and between ethicists and medical specialists.
Stakeholder Hearings
Stakeholder hearings were organized early to include all issues relevant to patients (comprehensiveness of the HTA) and to ensure public involvement in the process (Reference Street, Stafinski, Lopes and Menon16). One hearing was for NGOs (the Association for LGBTI Rights in Finland, Association for transgender and intersex rights and Association of the families of trans children and adolescents) and another for patients, who were recruited via GD clinics and could participate anonymously. The hearings were led by the ethicists.
The NGOs emphasized structural problems leading to poor access to services. Gatekeeping by medical professionals was seen as too strict, and treatment alternatives and reimbursement limited. NGOs emphasized the centrality of autonomy and interpreted this as individuals’ right to choose the treatments most suitable to their gender identity. In their view, empirical evidence for many treatments was convincing, and benefits generally outweighed the risks. The NGOs envisioned not only medical, but also legislative changes, supporting a free right to choose one’s legal gender.
Discussions with patients focused more on personal experiences. Transgender men and women were more satisfied with their experiences in the health system than nonbinary. Dysphoria, distress, and social prejudices during the evaluation and treatment phases were common. Lack of psychological support in the face of complex decisions with life-long consequences was reported, as was the importance of family support.
Stakeholder hearings provided important input for the HTA. The outcomes expected by patients turned out to be so complex, that the literature review was not limited to any predetermined set of outcomes. The importance of psychosocial support was emphasized in the report partly due to these hearings that clarified the strenuousness of the process from the patient perspective. Hearings also suggested that NGO discourse could be difficult to reconcile with the professional discourse. Understanding the complexity of combining these discourses helped workgroup members better understand the social context of the HTA.
Summary and Appraisal Phase
Balancing the Ethical Arguments
Discussions about ethical themes were formalized into ethical arguments by the ethicists, using ethical theories recommended by the EUnetHTA model. The ethical arguments were brought up for repeated discussions, clarifications, and consensus-building in the workgroup, before writing the ethics report. These discussions did not significantly change the ethical argumentation presented by the ethicists. Thus, although the EUnetHTA model separates data-gathering and balancing phases, in practice most of the discussions relevant for balancing the arguments were held already in the data-gathering phase.
Ethical theories used and key themes discussed were:
Casuistic approach proceeds from cases to a more general level. Problematic cases are compared to cases, where generally accepted solution exists. Coherence analysis emphasizes the importance of coherence between how different patient groups are treated, but also coherence between different discourses (9).
Casuistic analysis emphasized the need to identify the morally relevant differences between transgender, nonbinary, and cis-gender persons suffering from gender- or body-related dysphoria. A related issue discussed was whether transgender identity should be approached differently from nonbinary gender identities. Treatment of transgender men and women appeared fairly uncontroversial by patients and professionals (Reference Gerritse, Hartman and Antonides15). For nonbinary people, however, the desired outcomes of medical treatments do not necessarily match any clear reference category. This makes setting standards for adequate outcomes and, consequently, limits for public funding, difficult.
Another question related to funding raised by both casuistry and coherence analysis was, to what extent should people with transgender and nonbinary identities be treated differently from cis-gender people. Cis-gender individuals can be misgendered due to bodily appearance, but these problems are not usually publicly funded (exceptions include breast reconstructions after cancer surgery). This was not seen as relevant by the clinicians at first. However, the issue of breast reduction operations due to dysphoria or social consequences, irrespective of gender identity, was later repeatedly discussed.
Clinicians saw the practical importance of coherence, and the problems related to not having objective reference points. Unambiguous guideline stipulating the limits of publicly funded treatments was considered essential. It was understood that many decisions cannot be made based on empirical facts and evidence alone, for example, deciding how many body-hair-removal treatments and by what methods should be publicly funded. No clear principles for setting these limits could be conjured, so guidelines were crafted more on casuistic and coherentist grounds.
Coherence analysis also works on a more abstract level, considering the coherence among different levels of discourses. Ethicists clarified this as the challenge of combining the discourses of NGOs and professionals, or discourses based on public health policy, evidence-based medicine, and human rights. Ethicists emphasized the importance of coherence, to create a guideline that would be acceptable to all parties and thus sustainable. Public health point of view emphasizes the importance of treating different health problems equally and maximizing the cost-effectiveness of publicly funded treatments. Human rights view emphasizes individual right to self-determination and receiving treatments. Although these discourses can in theory be reconciled, this appeared difficult in practice regarding GD. One reason for this was that professionals saw real-life health-care resources as a zero-sum game, thus needing objective criteria to define the limits of publicly funded treatments. The unresolvability of these conflicting discourses was, in itself, an important result of the ethics discussions.
Principlism is a method that assumes certain ethical principles to exist, and that these principles should be followed, other things being equal. The EUnetHTA uses the most widely used model comprising of respect for autonomy, nonmaleficence, beneficence, and justice (Reference Beauchamp and Childress17). Most discussions regarding autonomy concerned adolescents. Ethicists and adolescent psychiatrists emphasized how autonomous decision making is likely to be difficult due to poor evidence base and general difficulty of predicting life-long consequences of options. The adolescent psychiatrists presented a fundamental problem regarding autonomy in the form of situations where gender identity had not stabilized when the person was seeking treatment. This may make the person incompetent to make decisions regarding permanent physical body modification treatments. Treatment decisions may also influence which way the gender identity of adolescents develops (Reference Kaltiala-Heino, Bergman, Työläjärvi and Frisen18). This makes noncontrolled follow-up studies potentially misleading.
The above discussion resurfaced when the balance of harms and benefits of treating adolescents was discussed. There appeared to be a fundamental difference in values regarding interpretation of evidence and the burden of proof: NGOs emphasized individual choice and right for informed risk-taking, whereas many professionals emphasized lack of evidence and risk of regret as reasons for not giving treatments.
A common conclusion of principlistic discussions was the importance of psychosocial evaluation and counseling. Counseling was seen by the workgroup as valuable in increasing autonomy, but also likely beneficial for treating dysphoria and concurrent mental health problems directly, with low probability of harms. This view was shared by the patients and NGOs in the hearings. When counseling was included in the recommendation, however, this was also criticized by the NGOs. Counseling at the first contact, and psychological evaluation, when necessary, prior to admittance to the university hospitals gender identity units, was seen as limiting patient autonomy and as unnecessary gatekeeping. This difference was interpreted as a consequence of NGO’s emphasis on psychosocial problems being secondary to gender identity issues, in contrast to professionals’ emphasis on first identifying and treating psychosocial problems related to other causes. These discussions on autonomy influenced the final recommendation, where improved access to psychosocial counseling was emphasized, along with a caution in recommending permanent treatments for adolescents.
Social shaping of technology (SST) is a view in which health technologies and treatments are assessed in a larger societal framework. Health technologies are used in a social context that affects their use, and technologies in turn affect society. For example, the changes in diagnostic classifications will be reflected on the social side. Interplay between individuals, society, and medical treatments available for GD is complex, and was not deeply studied in the process. Discussions about SST increased the understanding of role of the HTA, but also of the limits of medical treatments of GD. The outcome of these discussions was, that a separate chapter about the role of society was written. Medical interventions alone are not sufficient to improve the well-being of transgender people.
Public Commentary of the Report
The draft of the ethical analysis, and the whole HTA, was available online for comments for the general public before finalizing. Altogether sixty-nine commentaries were received. Some disagreements with the results and some useful comments regarding the recommendation came up, but none concerning the ethical report directly. No substantial alterations to the report were made based on this public commentary.
Discussion
This paper describes how an integrated ethical analysis using EUnetHTA model influenced a HTA concerning an exceptionally controversial and complex topic. The paper describes the key steps where ethical discussions were incorporated in the HTA process, summarizes the content of these discussions, and explicates the impact of these discussions on the process and final recommendation.
This paper also attempts, by extension, to show what kind of effects omitting ethical analysis can have. The ethical analysis emphasized at least the following themes that might otherwise have been foregone.
In the planning phase, ethics was essential in showing the complexity and social connectedness of the topic. GD is an unusually complex issue that cannot be understood without reference to social surroundings. Although professionals tended to see GD from their own specialty’s point of view, the ethicists were important in emphasizing the complex interplay between social, psychological, and biomedical aspects. This influenced how the HTA was scoped and executed and helped the HTA group work together on an emotionally charged topic.
In the evidence-gathering phase, at least two significant ethical themes emerged and influenced the results.
First, ethicists could formulate a nuanced view of autonomy, which brought structure and clarity to the conflicting views and intuitions different people had on the topic. The interpretation and discussions of autonomy turned out to be important for the recommendation, emphasizing caution on irreversible treatments of adolescents, and the importance of psychosocial support.
Second, justice and equality between different patient groups would not have received the same attention without the ethicists. Profession-related bias is well-known issue guideline work (Reference Gotzsche and Ioannidis19). Professionals treating only certain patient groups cannot easily compare and balance the problems and treatment needs of their own patients to other groups. The same is true of patient advocacy groups. Justice is fundamentally important for organizations like COHERE, that issue recommendations with direct resource implications. Ethicists, like health economists, lawyers, and information specialists, are important in emphasizing the justice, equality and coherence on the healthcare system level.
Toward the summary phase, the role of ethics shifted from presenting arguments toward facilitating coherence. Balancing different ethical arguments with explicit ethical theories helped in this, as did explaining how some disagreements between discourses might remain irresolvable. In a way, integrating ethics expertise was the key element that made the HTA an interactive exercise that aimed toward creating a recommendation that would be as valid and as acceptable as possible.
The key strength of this paper is that it describes a real-world HTA conducted in a governmental setting, where not only assessment but also appraisal and a clear recommendation was a mandatory outcome. The EUnetHTA model was used, but the results should be useful to users of other models too. The ethical issues identified are likely to be relevant to health policy discussions regarding GD also outside HTA setting. The weakness of the paper is its narrative format, so the exact effects of including an ethical analysis cannot be proved. This is, however, an inevitable consequence of the integrated approach to HTA and ethics, where ethical discussions are integrated in the HTA process from start to finish.
Our paper shows that EUnetHTA core model is a feasible method for integrating ethical analysis into an HTA, in an extensively complex topic like GD. Our experience also supports many of the theoretical ideas from INTEGRATE-HTA project regarding how different kinds of complexity in ethics could be approached (Reference Lysdahl, Oortwijn and van der Wilt20). First, plurality of perspectives was an important and difficult issue, but reasonably approached with stakeholder involvement and cooperation between all experts throughout the HTA. Uncertain causality, difficulty in determining the phenomena and unpredictable outcomes were noted early, as ethical considerations were included in the planning phase. The EUnetHTA core model’s highly structured question-based data collection phase functioned well in identifying ethical complexity and conflicting values. Thus, the first two phases of the EUnetHTA model functioned well: ethical issues were taken into account in the planning phase, and detailed ethical themes were systematically identified and discussed with content experts in the data-gathering phase. However, the analysis phase, where conflicting values should have been solved, functioned less satisfactorily. Using several ethical theories to analyze the issues was feasible for the ethicists but presenting all these analyses for the workgroup to discuss and reach consensus turned out to be prohibitively time-consuming.
In sum, our paper shows how an ethical analysis can be a valuable part of a HTA process, especially in complex and socially controversial topics. Including or omitting an ethical analysis can have a significant impact on the HTA process and resulting recommendations.
Supplementary material
To view supplementary material for this article, please visit https://doi.org/10.1017/S0266462322003257.
Acknowledgments
The authors gratefully acknowledge the contribution of all COHERE workgroup members who took part in preparing the HTA on which this paper is based.
Funding statement
The HTA and the original ethical analysis were funded by the Ministry of Social Affairs and Health, Finland. This work was supported by a grant from the Signe & Ane Gyllenberg Foundation (SS).
Conflicts of Interest
The authors declare none.
