Medicine in the marketplace: clinician and patient views on commercial influences on assisted reproductive technology.

RESEARCH QUESTION: What are the views and experiences of patient and expert stakeholders on the positive and negative impacts of commercial influences on the provision of assisted reproductive technology (ART) services, and what are their suggestions for governance reforms? DESIGN: Semi-structured interviews were conducted with 31 ART industry experts from across Australia and New Zealand and 25 patients undergoing ART from metropolitan and regional Australia, between September 2020 and September 2021. Data were analysed using thematic analysis. RESULTS: Expert and patient participants considered that commercial forces influence the provision of ART in a number of positive ways - increasing sustainability, ensuring consistency in standards and providing patients with greater choice. Participants also considered commercial forces to have a number of negative impacts, including increased costs to government and patients; the excessive use of interventions that lack sufficient evidence to be considered part of standard care; inadequately informed consent (particularly with regard to financial information); and threats to patient-provider relationships and patient-centred care. Participants varied in whether they believed that professional self-regulation is sufficient. While recognizing the benefits of commercial investment in healthcare, many considered that regulatory reforms, as well as organizational cultural initiatives, are needed as means to ensure the primacy of patient well-being. CONCLUSIONS: The views expressed in this study should be systematically and critically examined to derive insights into how best to govern ART. These insights may also inform the design and delivery of other types of healthcare that are provided in the private sector.

What are the anticipated benefits, risks, barriers and facilitators to implementing person-centred outcome measures into routine care for children and young people with life-limiting and life-threatening conditions? A qualitative interview study with key stakeholders.

BACKGROUND: There is a growing evidence-base underpinning implementation of person-centred outcome measures into adult palliative care. However evidence on how best to achieve this with children facing life-threatening and life-limiting conditions is limited. AIM: To identify the anticipated benefits, risks, barriers and facilitators to implementing person-centred outcome measures for children with life-limiting and life-threatening conditions. DESIGN: Cross-sectional qualitative semi-structured interview study with key stakeholders analysed using Framework analysis informed by the adapted-Consolidated Framework for Implementation Research. SETTING/PARTICIPANTS: A total of n = 26 children with life-limiting or life-threatening conditions, n = 40 parents/carers, n = 13 siblings and n = 15 health and social care professionals recruited from six hospitals and three children's hospices and n = 12 Commissioners of health services. RESULTS: All participants were supportive of future implementation of person-centred outcome measures into care. Anticipated benefits included: better understanding of patient and family priorities, improved communication and collaborative working between professionals and families and standardisation in data collection and reporting. Anticipated risks included increased workload for staff and measures not being used as intended. Implementation barriers included: acceptability and usability of outcome measures by children; burden and capacity of parents/carers regarding completion; privacy concerns; and language barriers. Implementation facilitators included designing measures using language that is meaningful to children and families, ensuring potential benefits of person-centred outcome measures are communicated to encourage 'buy-in' and administering measures with known and trusted professional. CONCLUSIONS: Implementation of person-centred outcome measures offer potential benefits for children with life-limiting and life-threatening conditions. Eight recommendations are made to maximise benefits and minimise risks in implementation.

Crafting representations of rare disease: collage as qualitative inquiry.

BACKGROUND: Collage is a modality of expression which involves repurposing and juxtaposing fragments. Our aim was to explore both how and what collage, as an arts-based research method, might contribute to enlivening understandings of the experiences of families affected by rare conditions. METHODS: During 10 weeks of collaging workshops participants created artistic representations of their experiences. The methodology produced a convivial atmosphere where participants talked openly about everyday challenges. RESULTS: The collages and conversations produced offer a means through which to consider the complex and multiple positions which families affected by rare disease interpolate. Particularly, the collages prompt cross-cutting thematic reflections on motherhood and care, the challenges of being heard, and balancing family life alongside medicalisation. CONCLUSIONS: The opportunity to convey topics and feelings through a medium which was both tentatively open yet conceptually complex allowed the broaching of sensitive and elusive themes in a safe, expressive, and non-threatening manner.

The ethical challenges of personalized digital health.

Personalized digital health systems (pHealth) bring together in sharp juxtaposition very different yet hopefully complementary moral principles in the shared objectives of optimizing health care and the health status of individual citizens while maximizing the application of robust clinical evidence through harnessing powerful and often complex modern data-handling technologies. Principles brought together include respecting the confidentiality of the patient-clinician relationship, the need for controlled information sharing in teamwork and shared care, benefitting from healthcare knowledge obtained from real-world population-level outcomes, and the recognition of different cultures and care settings. This paper outlines the clinical process as enhanced through digital health, reports on the examination of the new issues raised by the computerization of health data, outlines initiatives and policies to balance the harnessing of innovation with control of adverse effects, and emphasizes the importance of the context of use and citizen and user acceptance. The importance of addressing ethical issues throughout the life cycle of design, provision, and use of a pHealth system is explained, and a variety of situation-relevant frameworks are presented to enable a philosophy of responsible innovation, matching the best use of enabling technology with the creation of a culture and context of trustworthiness.

Ethical preparedness and developments in genomic healthcare.

Considerations of the notion of preparedness have come to the fore in the recent pandemic, highlighting a need to be better prepared to deal with sudden, unexpected and unwanted events. However, the concept of preparedness is also important in relation to planned for and desired interventions resulting from healthcare innovations. We describe ethical preparedness as a necessary component for the successful delivery of novel healthcare innovations, and use recent advances in genomic healthcare as an example. We suggest that practitioners and organisations charged with delivering innovative and ambitious healthcare programmes can only succeed if they are able to exhibit the attribute of ethical preparedness.

Spiritual, religious, and existential concerns of children and young people with life-limiting and life-threatening conditions: A qualitative interview study.

BACKGROUND: Despite being a core domain of palliative care, primary data on spiritual and existential concerns has rarely been collected among children with life-limiting and life-threatening conditions and their families. Existing evidence has tended to focus on the religious aspects among children with cancer. AIM: To identify the spiritual needs of children with life-limiting and life-threatening conditions. DESIGN: Cross-sectional semi-structured, qualitative interview study with children, families and health and social care professionals. Verbatim transcripts were analysed using Framework analysis. SETTING/PARTICIPANTS: Purposively sampled children with life-limiting and life-threatening conditions, their parents and siblings, health and social care professionals recruited from six hospitals and three children's hospices in the UK, and commissioners of paediatric palliative care services recruited through networks and a national charity. RESULTS: One hundred six participants were interviewed: 26 children (5-17 years), 53 family members (parents/carers of children 0-17 years and siblings (5-17 years)), 27 professionals (health and social care professionals and commissioners of paediatric palliative care). Themes included: living life to the fullest, meaning of life and leaving a legacy, uncertainty about the future, determination to survive, accepting or fighting the future and role of religion. Children as young as 5 years old identified needs or concerns in the spiritual domain of care. CONCLUSIONS: Addressing spiritual concerns is essential to providing child- and family-centred palliative care. Eliciting spiritual concerns may enable health and social care professionals to identify the things that can support and enhance a meaningful life and legacy for children and their families.

Composition and capacity of Institutional Review Boards, and challenges experienced by members in ethics review processes in Addis Ababa, Ethiopia: An exploratory qualitative study.

Few studies in sub-Saharan Africa evaluate Institutional Review Boards (IRBs) capacity. The study aims to explore the composition of IRBs, training, and challenges experienced in the ethics review processes by members of research institutions and universities in Addis Ababa, Ethiopia. Our findings indicate that most IRBs members were trained on research ethics and good clinical practice. However, majority perceived the trainings as basic. IRB members faced several challenges including: investigators wanting rapid review; time pressure; investigators not following checklists; limited expertise in reviewing clinical trials, studies on genetics, and traditional medicine; lack of IRB offices for administrative work; competing tasks; limited staffing and the lack of a standardized review system. There is need for advanced training on research ethics to meet the evolving research needs. In addition, investments in IRBs are needed in terms of funding, and physical and human resources in Addis Ababa and Ethiopia in general.

Achieving child-centred care for children and young people with life-limiting and life-threatening conditions-a qualitative interview study.

This study aims to identify the symptoms, concerns, and care priorities of children with life-limiting conditions and their families. A semi-structured qualitative interview study was conducted, seeking perspectives from multiple stakeholders on symptoms, other concerns, and care priorities of children and young people with life limiting and life-threatening conditions and their families. Participants were recruited from six hospitals and three children's hospices in the UK. Verbatim transcripts were analysed using framework analysis. A total of 106 participants were recruited: 26 children (5-17 years), 40 parents (of children 0-17 years), 13 siblings (5-17 years), 15 health and social care professionals, 12 commissioners. Participants described many inter-related symptoms, concerns, and care priorities impacting on all aspects of life. Burdensome symptoms included pain and seizures. Participants spoke of the emotional and social impacts of living with life-limiting conditions, such as being able to see friends, and accessing education and psychological support. Spiritual/existential concerns included the meaning of illness and planning for an uncertain future. Data revealed an overarching theme of pursuing 'normality', described as children's desire to undertake usual childhood activities. Parents need support with practical aspects of care to help realise this desire for normality. CONCLUSION: Children with life-limiting conditions and their families experience a wide range of inter-related symptoms, concerns, and care priorities. A holistic, child-centred approach to care is needed, allowing focus on pursuit of normal childhood activities. Improvements in accessibility, co-ordination, and availability of health services are required to achieve this. WHAT IS KNOWN: • Existing evidence regarding symptoms, concerns, and care priorities for children with life-limiting conditions is largely limited to proxy-reported data and those with a cancer diagnosis. • Child-centred care provision must be directed by children's perspectives on their priorities for care. WHAT IS NEW: • Social and educational activities are more important to children with life-limiting conditions than their medical concerns. • A holistic approach to care is required that extends beyond addressing medical needs, in order to support children with life-limiting conditions to focus on pursuit of normal childhood activities.

"I wanna live and not think about the future" what place for advance care planning for people living with severe multiple sclerosis and their families? A qualitative study.

BACKGROUND: Little is known about how people with multiple sclerosis (MS) and their families comprehend advance care planning (ACP) and its relevance in their lives. AIM: To explore under what situations, with whom, how, and why do people with MS and their families engage in ACP. METHODS: We conducted a qualitative study comprising interviews with people living with MS and their families followed by an ethical discussion group with five health professionals representing specialties working with people affected by MS and their families. Twenty-seven people with MS and 17 family members were interviewed between June 2019 and March 2020. Interviews and the ethical discussion group were audio-recorded and transcribed verbatim. Data were analysed using the framework approach. RESULTS: Participants' narratives focused on three major themes: (i) planning for an uncertain future; (ii) perceived obstacles to engaging in ACP that included uncertainty concerning MS disease progression, negative previous experiences of ACP discussions and prioritising symptom management over future planning; (iii) Preferences for engagement in ACP included a trusting relationship with a health professional and that information then be shared across services. Health professionals' accounts from the ethical discussion group departed from viewing ACP as a formal document to that of an ongoing process of seeking preferences and values. They voiced similar concerns to people with MS about uncertainty and when to initiate ACP-related discussions. Some shared concerns of their lack of confidence when having these discussions. CONCLUSION: These findings support the need for a whole system strategic approach where information about the potential benefits of ACP in all its forms can be shared with people with MS. Moreover, they highlight the need for health professionals to be skilled and trained in engaging in ACP discussions and where information is contemporaneously and seamlessly shared across services.

Writing the worlds of genomic medicine: experiences of using participatory-writing to understand life with rare conditions.

The diagnostic and treatment possibilities made possible by the development and subsequent mainstreaming of clinical genomics services have the potential to profoundly change the experiences of families affected by rare genetic conditions. Understanding the potentials of genomic medicine requires that we consider the perspectives of those who engage with such services; there are substantial social implications involved. There are increasing calls to think more creatively, and draw on more participatory approaches, in evoking rich accounts of lived experience. In this article, we discuss our rationale for, and experiences of, using 'participatory-writing' to understand the diverse, variable and multilayered everyday lives of families and how these correspond with the emerging, rapidly changing and complex field of genomic medicine. Participatory-writing has many benefits as a method for social inquiry. Writing can be expressive and self-revelatory, providing insight into personal and sensitive topics. Writing together produces new conversations and relationships. Pieces written by participants have the potential to affect readers, evoking and enlivening research and prompting professional change. Working with a writing tutor, we organised a participatory-writing programme for families touched by genetic conditions. This involved a series of workshops with an emphasis on building confidence in expressing lived experience through experimenting with different writing techniques. Afterwards we arranged reflective interviews with participants. We drew on dialogical narrative analysis to engage with participants' written pieces, and highlight what everyday life is like for the people who live with, and care for, those with genetic conditions. The stories produced through our writing-groups unfold the implications of new genomic technologies, illuminating how genomics acts to (and likewise, fails to) reconfigure aspects of people's lives outside of the clinic, while simultaneously existing as a sociotechnical frame that can eclipse the wider contexts, challenges and liveliness of life with rare genetic conditions.

COVID-19: Impact on Pediatric Palliative Care.

CONTEXT: Children and young people with life-limiting or life-threatening conditions and their families are potentially vulnerable during COVID-19 lockdowns due to pre-existing high clinical support needs and social participation limitations. OBJECTIVES: To explore the impact of the COVID-19 pandemic and lockdowns on this population. METHODS: Sub-analysis of an emergent COVID-19 related theme from a larger semi-structured interview study investigating priority pediatric palliative care outcomes. One hundred and six United Kingdom-wide purposively-sampled Children and young people with life-limiting or life-threatening conditions, parent/carers, siblings, health professionals, and commissioners. RESULTS: COVID-19 was raised by participants in 12/44 interviews conducted after the United Kingdom's first confirmed COVID-19 case. Key themes included loss of vital social support, disruption to services important to families, and additional psychological distress. CONCLUSION: Continued delivery of child- and family-centered palliative care requires innovative assessment and delivery of psycho-social support. Disruptions within treatment and care providers may compound support needs, requiring cordination for families facing multiagency delays.

Imagining genomic medicine futures in primary care: General practitioners' views on mainstreaming genomics in the National Health Service.

Genomic medicine has captured the imaginations of policymakers and medical scientists keen to harness its health and economic potentials. In 2012, the UK government launched the 100,000 Genomes Project to sequence the genomes of British National Health Service (NHS) patients, laying the ground for mainstreaming genomic medicine in the NHS and developing the UK's genomics industry. However, the recent research and reports from national bodies monitoring genomic medicine's roll-out suggest both ethical and practical challenges for health-care professionals. Against this backdrop, this paper, drawing on qualitative research interviews with general practitioners (GPs) and documentary analysis of policy, explores GPs' views on mainstreaming genomic medicine in the NHS and implications for their practice. Analysing the NHS's genomic medicine agenda as a 'sociotechnical imaginary', we demonstrate that whilst sociotechnical imaginaries are construed as collectively shared understandings of the future, official visions of genomic medicine diverge from those at the forefront of health-care service delivery. Whilst policy discourse evokes hope and transformation of health care, some GPs see technology in formation, an unattainable 'utopia', with no relevance to their everyday clinical practice. Finding space for genomics requires bridging the gap between 'work as imagined' at the policy level and 'work as done' in health-care delivery.

Ethics review of big data research: What should stay and what should be reformed?

BACKGROUND: Ethics review is the process of assessing the ethics of research involving humans. The Ethics Review Committee (ERC) is the key oversight mechanism designated to ensure ethics review. Whether or not this governance mechanism is still fit for purpose in the data-driven research context remains a debated issue among research ethics experts. MAIN TEXT: In this article, we seek to address this issue in a twofold manner. First, we review the strengths and weaknesses of ERCs in ensuring ethical oversight. Second, we map these strengths and weaknesses onto specific challenges raised by big data research. We distinguish two categories of potential weakness. The first category concerns persistent weaknesses, i.e., those which are not specific to big data research, but may be exacerbated by it. The second category concerns novel weaknesses, i.e., those which are created by and inherent to big data projects. Within this second category, we further distinguish between purview weaknesses related to the ERC's scope (e.g., how big data projects may evade ERC review) and functional weaknesses, related to the ERC's way of operating. Based on this analysis, we propose reforms aimed at improving the oversight capacity of ERCs in the era of big data science. CONCLUSIONS: We believe the oversight mechanism could benefit from these reforms because they will help to overcome data-intensive research challenges and consequently benefit research at large.

A realist review of advance care planning for people with multiple sclerosis and their families.

BACKGROUND: Advance care planning (ACP) is reported to improve the quality of outcomes of care among those with life-limiting conditions. However, uptake is low among people living with multiple sclerosis (MS) and little is known about why or how people with MS engage in this process of decision-making. AIMS: To develop and refine an initial theory on engagement in ACP for people with MS and to identify ways to improve its uptake for those who desire it. METHODS: Realist review following published protocol and reporting following Realist and Meta-narrative Evidence Synthesis: Evolving Standards (RAMESES) guidelines. A multi-disciplinary team searched MEDLINE, PsychInfo, CINAHL, Scopus, Web of Science, Embase, Google Scholar in addition to other sources from inception to August 2019. Quantitative or qualitative studies, case reports, and opinion or discussion articles related to ACP and/or end of life discussions in the context of MS were included, as well as one article on physical disability and one on motor neuron disease, that contributed important contextual information. Researchers independently screened abstracts and extracted data from full-text articles. Using abductive and retroductive analysis, each article was examined for evidence to support or refute 'context, mechanism, and outcome' (CMO) hypotheses, using the Integrated Behaviour Model to guide theory development. Quality was assessed according to methodological rigour and relevance of evidence. Those studies providing rich descriptions were synthesised using a realist matrix to identify commonalities across CMO configurations. RESULTS: Of the 4,034 articles identified, 33 articles were included in the synthesis that supported six CMO hypotheses that identified contexts and mechanisms underpinning engagement in ACP for people with MS and included: acceptance of their situation, prior experiences, confidence, empowerment, fear (of being a burden, of death and of dying) and the desire for autonomy. Acceptance of self as a person with a life-limiting illness was imperative as it enabled people with MS to see ACP as pertinent to them. We identified the context of MS-its long, uncertain disease trajectory with periods of stability punctuated by crisis-inhibited triggering of mechanisms. Similarly, the absence of skills and confidence in advanced communication skills among health professionals prevented possibilities for ACP discussions taking place. CONCLUSION: Although mechanisms are inhibited by the context of MS, health professionals can facilitate greater uptake of ACP among those people with MS who want it by developing their skills in communication, building trusting relationships, sharing accurate prognostic information and sensitively discussing death and dying.

Towards a national genomics medicine service: the challenges facing clinical-research hybrid practices and the case of the 100 000 genomes project.

Clinical practice and research are governed by distinct rules and regulations and have different approaches to, for example, consent and providing results. However, genomics is an example of where research and clinical practice have become codependent. The 100 000 genomes project (100kGP) is a hybrid venture where a person can obtain a clinical investigation only if he or she agrees to also participate in ongoing research-including research by industry and commercial companies. In this paper, which draws on 20 interviews with professional stakeholders involved in 100kGP, we investigate the ethical issues raised by this project's hybrid nature. While some interviewees thought the hybrid nature of 100kGP was its vanguard, interviewees identified several tensions around hybrid practice: how to decide who should be able to participate; how to determine whether offering results might unduly influence participation into wide-ranging but often as yet unknown research and how to ensure that patients/families do not develop false expectations about receiving results. These areas require further debate as 100kGP moves into routine healthcare in the form of the national genomic medicine service. To address the tensions identified, we explore the appropriateness of Faden et al.'s framework of ethical obligations for when research and clinical care are completely integrated. We also argue that enabling ongoing transparent and trustworthy communication between patients/families and professionals around the kinds of research that should be permitted in 100kGP will help to understand and ensure that expectations remain realistic. Our paper aims to encourage a focused discussion about these issues and to inform a new 'social contract' for research and clinical care in the health service.

Genomics England's implementation of its public engagement strategy: Blurred boundaries between engagement for the United Kingdom's 100,000 Genomes project and the need for public support.

The United Kingdom's 100,000 Genomes Project has the aim of sequencing 100,000 genomes from National Health Service patients such that whole genome sequencing becomes routine clinical practice. It also has a research-focused goal to provide data for scientific discovery. Genomics England is the limited company established by the Department of Health to deliver the project. As an innovative scientific/clinical venture, it is interesting to consider how Genomics England positions itself in relation to public engagement activities. We set out to explore how individuals working at, or associated with, Genomics England enacted public engagement in practice. Our findings show that individuals offered a narrative in which public engagement performed more than one function. On one side, public engagement was seen as 'good practice'. On the other, public engagement was presented as core to the project's success - needed to encourage involvement and ultimately recruitment. We discuss the implications of this in this article.

Public trust and 'ethics review' as a commodity: the case of Genomics England Limited and the UK's 100,000 genomes project.

The UK Chief Medical Officer's 2016 Annual Report, Generation Genome, focused on a vision to fully integrate genomics into all aspects of the UK's National Health Service (NHS). This process of integration, which has now already begun, raises a wide range of social and ethical concerns, many of which were discussed in the final Chapter of the report. This paper explores how the UK's 100,000 Genomes Project (100 kGP)-the catalyst for Generation Genome, and for bringing genomics into the NHS-is negotiating these ethical concerns. The UK's 100 kGP, promoted and delivered by Genomics England Limited (GEL), is an innovative venture aiming to sequence 100,000 genomes from NHS patients who have a rare disease, cancer, or an infectious disease. GEL has emphasised the importance of ethical governance and decision-making. However, some sociological critique argues that biomedical/technological organisations presenting themselves as 'ethical' entities do not necessarily reflect a space within which moral thinking occurs. Rather, the 'ethical work' conducted (and displayed) by organisations is more strategic, relating to the politics of the organisation and the need to build public confidence. We set out to explore whether GEL's ethical framework was reflective of this critique, and what this tells us more broadly about how genomics is being integrated into the NHS in response to the ethical and social concerns raised in Generation Genome. We do this by drawing on a series of 20 interviews with individuals associated with or working at GEL.

The UK's 100,000 Genomes Project: manifesting policymakers' expectations.

The UK's 100,000 Genomes Project has the aim of sequencing 100,000 genomes from UK National Health Service (NHS) patients while concomitantly transforming clinical care such that whole genome sequencing becomes routine clinical practice in the UK. Policymakers claim that the project will revolutionize NHS care. We wished to explore the 100,000 Genomes Project, and in particular, the extent to which policymaker claims have helped or hindered the work of those associated with Genomics England - the company established by the Department of Health to deliver the project. We interviewed 20 individuals linked to, or working for Genomics England. Interviewees had double-edged views about the context within which they were working. On the one hand, policymakers' expectations attached to the venture were considered vacuous "genohype"; on the other hand, they were considered the impetus needed for those trying to advance genomic research into clinical practice. Findings should be considered for future genomes projects.