Coping and end-of-life decision-making in ALS: A qualitative interview study.

How do people with amyotrophic lateral sclerosis (PALS) deal with their diagnosis and engage in end-of-life decision-making? What informational or supportive needs do they have for counselling about life-sustaining treatment and end-of-life care? Which correlating conditions and influences relate to these needs and how do they connect to the wish to die or wish to live? We conducted a qualitative interview study with 13 people with ALS in Germany from March 2019 to April 2021. Data collection and analysis followed a grounded theory-based approach and revealed close relationships between coping, informational needs and the preparedness for decision-making. We identified the coping strategies 'avoid thinking about end-of-life' and its counterpart, 'planning ahead to be well-prepared,' and differentiated the latter into the patterns 'withdrawing from life and taking precautions against life-prolongation' and 'searching for a new meaning in life and preparing for life-sustaining treatment'. The approaches are based on individual perceptions, attitudes and motives and can be positively/negatively reinforced by healthcare professionals (HCP), family and other interpersonal networks, but also by disease progression and in reaction to health care services. Type and degree of needs concerning information and counselling differed according to coping strategies. These strategies may vary over time, resulting in different support needs. Our findings signify that deep insight is needed into PALS' coping processes to understand their decision-making about life-sustaining treatment. Healthcare professionals should be sensitive to illness experiences beyond medical aspects and foster coping as a biographical process to better support people with ALS.

Talking about the end of life: communication patterns in amyotrophic lateral sclerosis - a scoping review.

Amyotrophic lateral sclerosis (ALS) leads to death on average 2-4 years after the onset of symptoms. Although many people with the disease decide in favour of life-sustaining measures, some consider hastening death. The objectives of this review are to provide an insight into the following questions: (1) How do people with amyotrophic lateral sclerosis (PALS), their families and health care professionals (HCPs) communicate about life-sustaining and life-shortening options? (2) What are the challenges for all involved in decision making and communication about this topic? To answer these questions, we searched eight databases for publications in English and German on end-of-life issues of PALS. We included texts published between 2008 and 2018, and updated our search to May 2020. Sources were analysed in MAXQDA using deductively and inductively generated codes. After the final analysis, 123 full texts were included in this review. We identified a wide range of communicative challenges and six different and, in part, opposite communication patterns: avoiding or delaying communication on end-of-life issues, openly considering dying and actively seeking assistance, ignoring or disregarding patients' wishes, discussing and respecting the patients' wishes, engaging in advance care planning and avoiding or delaying advance care planning. The literature reveals a very heterogeneous response to end-of-life issues in ALS, despite several good-practice suggestions, examples and guidelines. We derive a strong need for harmonization and quality assurance concerning communication with PALS. Avoiding or delaying communication, decision making and planning, as well as ignoring or disregarding the patient's will by HCP can be judged as a violation of the ethical principles of autonomy and non-maleficence.

The wish to die and hastening death in amyotrophic lateral sclerosis: A scoping review.

BACKGROUND: Amyotrophic lateral sclerosis (ALS) develops into a life-threatening condition 2 to 4 years after the onset of symptoms. Although many people with the disease decide in favour of life-sustaining measures, thoughts about hastening death are not uncommon. OBJECTIVES: Our aim was to examine the scope of literature on the wish to die in ALS and provide an insight into determinants and motives for different end-of-life options. METHODS: We searched eight databases for English and German publications on death wishes in ALS for the period from 2008 to 2018 and updated the search up to May 2020. After the screening process, 213 full texts were included for the final analysis. We analysed the texts in MAXQDA, using deductively and inductively generated codes. RESULTS: We identified end-of-life considerations, ranging from wishes to die without hastening death, to options with the possibility or intention of hastening death. Besides physical impairment, especially psychosocial factors, socio-demographic status and socio-cultural context have a great impact on decisions for life-shortening options. There is huge variation in the motives and determinants for end-of-life considerations between individuals, different societies, healthcare and legal systems. CONCLUSIONS: For a variety of reasons, the information and counselling provided on different options for sustaining life or hastening death is often incomplete and insufficient. Since the motives and determinants for the wish to hasten death are extremely diverse, healthcare professionals should investigate the reasons, meaning and strength of the desire to die to detect unmet needs and examine which interventions are appropriate in each individual case.

[Ethics University on Regenerative Medicine - As an Instrument for the Development of Valid Public Opinions?] / Ethik-Universität zur Regenerativen Medizin ­ ein Instrument der fundierten Meinungsbildung für Laien?

OBJECTIVES: In 2016, we invited interested citizens to participate in the "ethics university on regenerative medicine" at Hannover Medical School. The present study analyses if and how this discursive and informative event inspired participants to form their own opinion on the issues at hand and to develop their general ethics literacy. METHODS: The "ethics university" was performed twice in 2016; each run consisted of four single consecutive events. Lectures were combined with interactive learning stations, and group discussions. Opinions and information level of all participants were surveyed by means of a postal questionnaire before and after the course to detect any changes of opinions and information levels; additionally, we surveyed participants' self-assessment. Participants of the second run were asked to form a waiting list control group to compare results from first run-participants. Furthermore, we conducted a content analysis of group discussions during the ethics university. RESULTS: Of 168 participants of both runs, 101 took part in the pre/post-survey. In addition, 30 questionnaires of the waiting list control group were analysed. Participants showed a higher level of information after the ethics university (changes between 0.75 and 1.93 points on a five-point scale). Between 50.5 and 66.0% of participants indicated that their opinion on different issues had become either more affirmative or more disapprobative as a result of attending the ethics university. On average, opinions were more positive after participation (between 0.44 and 1.0 points on a 5-point scale). Respondents in the waiting list control group showed no changes in opinion or information level. Participants themselves felt that they formed their opinions mainly on the basis of information they received in lectures, conversations with experts, interactive learning sessions, and written information. However, for many participants, interacting with other participants in the group discussions, as well as reflecting their own views was an important to forming informed opinions. CONCLUSION: Results of the evaluation show that participants were inspired to form their own opinions by the ethics university and to develop their ethics literacy (e. g. ability to reflect on normative questions). For future ethics universities, the group of participants should be as diverse as possible. In addition, interactive and discursive elements should be given a higher priority.

Do Public Involvement Activities in Biomedical Research and Innovation Recruit Representatively? A Systematic Qualitative Review.

BACKGROUND: Public involvement activities (PIAs) may contribute to the governance of ethically challenging biomedical research and innovation by informing, consulting with and engaging the public in developments and decision-making processes. For PIAs to capture a population's preferences (e.g. on issues in whole genome sequencing, biobanks or genome editing), a central methodological requirement is to involve a sufficiently representative subgroup of the general public. While the existing literature focusses on theoretical and normative aspects of 'representation', this study assesses empirically how such considerations are implemented in practice. It evaluates how PIA reports describe representation objectives, the recruitment process and levels of representation achieved. METHODS: PIA reports were included from a systematic literature search if they directly reported a PIA conducted in a relevant discipline such as genomics, biobanks, biotechnology or others. PIA reports were analyzed with thematic text analysis. The text analysis was guided by an assessment matrix based on PIA-specific guidelines and frameworks. RESULTS: We included 46 relevant reports, most focusing on issues in genomics. 27 reports (59%) explicitly described representation objectives, though mostly without adjusting eligibility criteria and recruiting methods to the specific objective. 11 reports (24%) explicitly reported to have achieved the intended representation; the rest either reported failure or were silent on this issue. CONCLUSION: Representation of study samples in PIAs in biomedical research and innovation is currently not reported systematically. Improved reporting on representation would not only improve the validity and value of PIAs, but could also contribute to PIA results being used more often in relevant policy and decision-making processes.

Ethics Literacy and "Ethics University": Two Intertwined Models for Public Involvement and Empowerment in Bioethics.

BACKGROUND: Informing lay citizens about complex health-related issues and their related ethical, legal, and social aspects (ELSA) is one important component of democratic health care/research governance. Public information activities may be especially valuable when they are used in multi-staged processes that also include elements of information and deliberation. OBJECTIVES: This paper presents a new model for a public involvement activity on ELSA (Ethics University) and evaluation data for a pilot event. METHODS: The Ethics University is structurally based on the "patient university," an already established institution in some German medical schools, and the newly developed concept of "ethics literacy." The concept of "ethics literacy" consists of three levels: information, interaction, and reflection. The pilot project consisted of two series of events (lasting 4 days each). RESULTS: The thematic focus of the Ethics University pilot was ELSA of regenerative medicine. In this pilot, the concept of "ethics literacy" could be validated as its components were clearly visible in discussions with participants at the end of the event. The participants reacted favorably to the Ethics University by stating that they felt more educated with regard to the ELSA of regenerative medicine and with regard to their own abilities in normative reasoning on this topic. CONCLUSION: The Ethics University is an innovative model for public involvement and empowerment activities on ELSA theoretically underpinned by a concept for "ethics literacy." This model deserves further refinement, testing in other ELSA topics and evaluation in outcome research.

Current practice of public involvement activities in biomedical research and innovation: a systematic qualitative review.

BACKGROUND: A recent report from the British Nuffield Council on Bioethics associated 'emerging biotechnologies' with a threefold challenge: 1) uncertainty about outcomes, 2) diverse public views on the values and implications attached to biotechnologies and 3) the possibility of creating radical changes regarding societal relations and practices. To address these challenges, leading international institutions stress the need for public involvement activities (PIAs). The objective of this study was to assess the state of PIA reports in the field of biomedical research. METHODS: PIA reports were identified via a systematic literature search. Thematic text analysis was employed for data extraction. RESULTS: After filtering, 35 public consultation and 11 public participation studies were included in this review. Analysis and synthesis of all 46 PIA studies resulted in 6 distinguishable PIA objectives and 37 corresponding PIA methods. Reports of outcome translation and PIA evaluation were found in 9 and 10 studies respectively (20% and 22%). The paper presents qualitative details. DISCUSSION: The state of PIAs on biomedical research and innovation is characterized by a broad range of methods and awkward variation in the wording of objectives. Better comparability of PIAs might improve the translation of PIA findings into further policy development. PIA-specific reporting guidelines would help in this regard. The modest level of translation efforts is another pointer to the "deliberation to policy gap". The results of this review could inform the design of new PIAs and future efforts to improve PIA comparability and outcome translation.

International requirements for consent in biobank research: qualitative review of research guidelines.

Several stakeholders in the field of biobank research are currently developing and evaluating innovative consent procedures, and in doing so refer to national and international guidelines that regulate biomedical research. The objectives of this study were to present (1) a synthesis of all consent issues mentioned or required in guidelines that are of potential relevance to biobank research and (2) a detailed overview of similarities and differences between these guidelines. This analysis considered a purposive sample of the nine most internationally recognised research guidelines plus a national checklist for developing consent forms. The synthesis of all included research guidelines found 41 issues of potential relevance to consent procedures in biobank research. The guidelines differ substantially with respect to (1) how comprehensively they address these consent issues, (2) their definition and explanation of the issues and (3) how directly or indirectly the issues are mentioned. The set of 41 consent issues presented in this paper should be further developed in a continuous process. Our findings on the differences among major research guidelines also raise the issue of a more systematic development and revision process for these guidelines. The presented set of content issues potentially relevant to consent in biobank research can inform the balanced development or evaluation of consent forms, as well as the development and revision of corresponding research guidelines.

Practice variation across consent templates for biobank research. a survey of German biobanks.

INTRODUCTION: Informed, voluntary, and valid consent from biomaterial donors is a precondition for biobank research. Valid consent protects donors' rights and helps maintain public trust in biobank research. Harmonization of consent procedures in biobank research is needed, because of the widely shared vision on national and international networking of biobanks including data and sample sharing. So far, no study has assessed and compared the content of current consent forms especially for biobank research. The objective of this study was to perform a content analysis of consent forms in German biobanks. METHODS: Based on ten guidelines for biomedical research, we developed an assessment matrix with 41 content issues that are potentially relevant for consent forms in biobank research. This assessment matrix was applied in a thematic text analysis to 30 consent documents of German biobanks identified via the German Biobank Registry in July 2012. RESULTS: Coverage of the 41 items in the assessed consent forms varied widely. For example, the items "Right to withdraw consent (without disadvantage)," "Policy for genetic information/consent to genetic analyzes" and "International cooperation/transborder use" were addressed in 97, 40, and 23% of all 30 consent forms respectively. The number of items covered by a single consent form ranged from 9 to 36 (22-88% out of 41 items). DISCUSSION: Our findings serve as a starting point to reflect upon the spectrum of consent issues that must be addressed in biobank research. The findings show that the majority of consent forms for German biobanks, if not all, should be improved and harmonized to better support an informed and balanced choice of potential donors and to facilitate networking of biobanks. Best practice models for consent forms in biobank research should be developed and biobank operators need to be more aware of relevant consent issues.

Evidence-based health information from the users' perspective--a qualitative analysis.

BACKGROUND: Evidence-based information is a precondition for informed decision-making and participation in health. There are several recommendations and definitions available on the generation and assessment of so called evidence-based health information for patients and consumers (EBHI). They stress the importance of objectively informing people about benefits and harms and any uncertainties in health-related procedures. There are also studies on the comprehensibility, relevance and user-friendliness of these informational materials. But to date there has been little research on the perceptions and cognitive reactions of users or lay people towards EBHI. The aim of our study is to define the spectrum of consumers' reaction patterns to written EBHI in order to gain a deeper understanding of their comprehension and assumptions, as well as their informational needs and expectations. METHODS: This study is based on an external user evaluation of EBHI produced by the German Institute for Quality and Efficiency in Health Care (IQWiG), commissioned by the IQWiG. The EBHI were examined within guided group discussions, carried out with lay people. The test readers' first impressions and their appraisal of the informational content, presentation, structure, comprehensibility and effect were gathered. Then a qualitative text analysis of 25 discussion transcripts involving 94 test readers was performed. RESULTS: Based on the qualitative text analysis a framework for reaction patterns was developed, comprising eight main categories: (i) interest, (ii) satisfaction, (iii) reassurance and trust, (iv) activation, (v) disinterest, (vi) dissatisfaction and disappointment, (vii) anxiety and worry, (viii) doubt. CONCLUSIONS: Many lay people are unfamiliar with core characteristics of this special information type. Two particularly critical issues are the description of insufficient evidence and the attendant absence of clear-cut recommendations. Further research is needed to examine strategies to explain the specific character of EBHI so as to minimize unintended or adverse reaction patterns. The presented framework describes the spectrum of users' reaction patterns to EBHI. It may support existing best practice models for editing EBHI.