SEMA6B variants cause intellectual disability and alter dendritic spine density and axon guidance.

Intellectual disability (ID) is a neurodevelopmental disorder frequently caused by monogenic defects. In this study, we collected 14 SEMA6B heterozygous variants in 16 unrelated patients referred for ID to different centers. Whereas, until now, SEMA6B variants have mainly been reported in patients with progressive myoclonic epilepsy, our study indicates that the clinical spectrum is wider and also includes non-syndromic ID without epilepsy or myoclonus. To assess the pathogenicity of these variants, selected mutated forms of Sema6b were overexpressed in Human Embryonic Kidney 293T (HEK293T) cells and in primary neuronal cultures. shRNAs targeting Sema6b were also used in neuronal cultures to measure the impact of the decreased Sema6b expression on morphogenesis and synaptogenesis. The overexpression of some variants leads to a subcellular mislocalization of SEMA6B protein in HEK293T cells and to a reduced spine density owing to loss of mature spines in neuronal cultures. Sema6b knockdown also impairs spine density and spine maturation. In addition, we conducted in vivo rescue experiments in chicken embryos with the selected mutated forms of Sema6b expressed in commissural neurons after knockdown of endogenous SEMA6B. We observed that expression of these variants in commissural neurons fails to rescue the normal axon pathway. In conclusion, identification of SEMA6B variants in patients presenting with an overlapping phenotype with ID and functional studies highlight the important role of SEMA6B in neuronal development, notably in spine formation and maturation and in axon guidance. This study adds SEMA6B to the list of ID-related genes.

Identifying, categorising, and mapping actors involved in resilience in healthcare: a qualitative stakeholder analysis.

BACKGROUND: Resilience in healthcare is the capacity to adapt to challenges and changes to maintain high-quality care across system levels. While healthcare system stakeholders such as patients, informal carers, healthcare professionals and service managers have all come to be acknowledged as important co-creators of resilient healthcare, our knowledge and understanding of who, how, and in which contexts different stakeholders come to facilitate and support resilience is still lacking. This study addresses gaps in the research by conducting a stakeholder analysis to identify and categorise the stakeholders that are key to facilitating and sustaining resilience in healthcare, and to investigate stakeholder relationships relevant for the enactment of resilient healthcare systems. METHODS: The stakeholder analysis was conducted using a sample of 19 empirical research projects. A narrative summary was written for 14 of the projects, based on publicly available material. In addition, 16 individual interviews were undertaken with researchers from the same sample of 19 projects. The 16 interview transcripts and 14 narratives made up the data material of the study. Application of stakeholder analysis methods was done in three steps: a) identification of stakeholders; b) differentiation and categorisation of stakeholders using an interest/influence grid; and c) investigation and mapping of stakeholder relationships using an actor-linkage matrix. RESULTS: Identified stakeholders were Patients, Family Carers, Healthcare Professionals, Ward/Unit Managers, Service or Case Managers, Regulatory Investigators, Policy Makers, and Other Service Providers. All identified stakeholders were categorised as either 'Subjects', 'Players', or 'Context Setters' according to their level of interest in and influence on resilient healthcare. Stakeholder relationships were mapped according to the degree and type of contact between the various groups of stakeholders involved in facilitating resilient healthcare, ranging from 'Not linked' to 'Fully linked'. CONCLUSION: Family carers and healthcare professionals were found to be the most active groups of stakeholders in the enactment of healthcare system resilience. Patients, managers, and policy makers also contribute to resilience to various degrees. Relationships between stakeholder groups are largely characterised by communication and coordination, in addition to formal collaborations where diverse actors work together to achieve common goals.

Conducting Social Science Research During Epidemics and Pandemics: Lessons Learnt.

The COVID-19 pandemic has had a significant impact on how field-based research is being conducted globally. Given the challenges of undertaking fieldwork during epidemics and the need for mixed methods research to address the social, political, and economic issues related to epidemics, there is a small but growing body of evidence in this area. To contribute to the logistical and ethical considerations for conducting research during a pandemic, we draw on the challenges and lessons learnt from adapting methods for two research studies conducted in 2021 during the COVID-19 pandemic in low- and middle-income country (LMIC) settings: (1) in-person research in Uganda and (2) combined remote and in-person research in South and Southeast Asia. Our case studies focus on data collection and demonstrate the feasibility of conducting mixed methods research, even with many logistical and operational constraints. Social science research is often used to identify the context of specific issues, to provide a needs assessment, or inform longer-term planning; however, these case studies have shown the need to integrate social science research from the start of a health emergency and in a systematic way. Social science research during future health emergencies can also inform public health responses during the emergency. It is also crucial to collect social science data after health emergencies to inform future pandemic preparedness. Finally, researchers need to continue research on other public health issues that are ongoing even during a public health emergency.

Researcher and study participants' perspectives of consent in clinical studies in four referral hospitals in Vietnam.

BACKGROUND: Within the research community, it is generally accepted that consent processes for research should be culturally appropriate and tailored to the context, yet researchers continue to grapple with what valid consent means within specific stakeholder groups. In this study, we explored the consent practices and attitudes regarding essential information required for the consent process within hospital-based trial communities from four referral hospitals in Vietnam. METHODS: We collected surveys from and conducted semi-structured interviews with study physicians, study nurses, ethics committee members, and study participants and family members regarding their experiences of participating in research, their perspectives toward research, and their views about various elements of the consent process. RESULTS: In our findings, we describe three interrelated themes related to the consent process: (1) words and regulation; (2) reimbursement, suspicions, and joining; and (3) responsibilities. In general, stakeholders had highly varied perspectives of nghiên cuu (Eng.: research) and researchers used varying levels of detail regarding all aspects of the study in the consent process to build trust with and/or promote potential research participants' choices about taking part in research. Findings additionally highlight how researchers felt that offering financial reimbursements in a hospital setting, where payment for services was routine, would be unfamiliar to participants and could raise suspicions about the research. Participants, however, focused their discussions on reimbursement or alternative reasons for joining the study, such as health related benefits or altruism. Finally, participants often relied on their physician to help them decide about joining a study or not. CONCLUSION: Further research is needed to understand how researchers and participants make sense of and practice consent, and how that impacts participants' decision-making about research participation. To promote valid consent within this context, it is important to engage with hospital-based trial communities as a whole. The data from this study will inform future research on consent, guide the revisions of consent related policies within our research sites and point to several larger issues surrounding researcher-participant expectations, communication, and trust.

The Therapeutic Engagement Questionnaire (TEQ): a service user-focused mental health nursing outcome metric.

BACKGROUND: Therapeutic engagement (TE) has been described as the crux of mental health nursing but despite its perceived importance, to date, there is no measurement tool that captures it. As a result, there is no way of determining the contribution of mental health nursing interaction to service user recovery, in acute inpatient mental health settings or the wider care quality agenda. METHODS: To develop and validate a TE measurement tool in partnership with Service Users (SUs) and Registered Mental Health Nurses (RMHNs). The TEQ was developed in 3 stages: 1) item generation (and pre-testing), 2) item reduction using Principal Component Analysis (PCA), and 3) validation across Mental Health Trusts in England. RESULTS: The final questionnaire has two versions, (SU and RMHN version), each scored within two contexts (1-1 SU-RMHN interactions and overall environment and atmosphere of the ward) and includes 20 items with two sub-scales (care interactions and care delivery). Psychometric evaluation of the TEQ shows high inter-scale correlations (0.66-0.95 SU; 0.57-0.90 RMHN), sound sub-scale internal consistency (> 0.95), concurrent validity (> 0.60) and adequate score variability for both versions of the TEQ. In summary, the TEQ behaves well as a measurement tool. CONCLUSIONS: The TEQ can determine the collaborative and empathic nature of RMHN-SU interactions, capture if SUs are treated with dignity and respect and recognise that the principles of the recovery approach are being respected. The TEQ can also provide robust monitoring of nursing activity, offer opportunity for transparency of activity, feed into healthcare organizations' key performance indicators and provide reassurance about the nature and quality of nurses' work.

Co-production for service improvement: Developing a training programme for mental health professionals to enhance medication adherence in Black, Asian and Minority Ethnic Service Users.

AIM: To co-produce consensus on the key issues important in educating mental health-care professionals to optimize mental health medication adherence in Black, Asian and Minority Ethnic (BAME) groups. OBJECTIVES: To identify perceptions of factors enabling or disabling medication adherence. To achieve consensus on content and delivery of an educational intervention for mental health-care professionals. METHODS: Data were collected from 2016 to 2018. Using individual interviews and a consensus workshop with carers and service users (SUs treated under the 1983 Mental Health Act 1983/revised 2007 for England and Wales), the experience of taking prescribed mental health medication and perspectives on adherence were explored. Data were analysed using 2-stage qualitative coding via the software tool NVivo version 11 to analyse transcribed data and to produce the main explanatory categories. RESULTS: SU and carer participants' perspectives substantially altered the original research design. The need to educate students rather than trained professionals was emphasized, and they suggested that educational content should be packaged in a contemporary manner (a virtual reality experience). Findings indicated that education should focus upon understanding the impact of taking prescribed antipsychotic medication on both SUs and carers. DISCUSSION: The importance of effective communication between health professionals, SUs and carers and a willingness to learn about and appreciate how BAME culture influences perception of mental illness and mental well-being were highlighted. CONCLUSION: In working co-productively, researchers need to be flexible and adaptable to change.

Whose voices? Patient and public involvement in clinical commissioning.

AIM: This paper aims to explore patient and public representation in a NHS clinical commissioning group and how this is experienced by staff and lay members involved. BACKGROUND: Patient and public involvement is believed to foster greater public representativeness in the development and delivery of health care services. However, there is widespread debate about what representation is or what it should be. Questions arise about the different constructions of representation and the representativeness of patients and the public in decision-making structures and processes. DESIGN: Ethnographic, two-phase study involving twenty-four observations across two types of clinical commissioning group meetings with patient and public involvement, fourteen follow-up interviews with NHS staff and lay members, and a focus group with five lay members. RESULTS: Perceptions of what constitutes legitimate representativeness varied between respondents, ranging from representing an individual patient experience to reaching large numbers of people. Consistent with previous studies, there was a lack of clarity about the role of lay members in the work of the clinical commissioning group. CONCLUSIONS: Unlike previous studies, it was lay members, not staff, who raised concerns about their representativeness and legitimacy. Although the clinical commissioning group provides resources to support patient and public involvement, there continues to be a lack of clarity about roles and scope for impact. Lay members are still some way from constituting a powerful voice at the table.

Service user involvement in the coproduction of a mental health nursing metric: The Therapeutic Engagement Questionnaire.

Service users' involvement in mental health service research is increasingly acknowledged as important, yet, whilst involving users of mental health services as research participants is commonplace, seeking out their experience and indeed their "expertise" to facilitate the development of tools to be used within mental health services is in its infancy. This article describes the involvement and views of service users in the development of a nursing metric-the Therapeutic Engagement Questionnaire. It presents their role in the three stages of development: generation, statement reduction and authentication.

Service user involvement in mental health care: an evolutionary concept analysis.

BACKGROUND: The concept of service user involvement is an evolving concept in the mental health-care literature. OBJECTIVE: This study sought to explore and analyse the concept of service user involvement as used in within the field of mental health care. METHODOLOGICAL APPROACH: An evolutionary concept analysis was conducted using a literature-based sample extracted from an electronic database search. One hundred and thirty-four papers met the inclusion criteria and were analysed to discover key attributes, antecedents and consequences of service user involvement and to produce a definition of the concept. FINDINGS: Five key attributes of service user involvement within the context of mental health care were identified: a person-centred approach, informed decision making, advocacy, obtaining service user views and feedback and working in partnership. DISCUSSION AND CONCLUSIONS: Clarity of the attributes and definition of the concept of service user involvement aims to promote understanding of the concept among key stakeholders including mental health professionals, service users and community and voluntary organizations. The findings of the research have utility in the areas of theory and policy development, research on service user involvement in mental health care and service user involvement in mental health practice. Directions for further research regarding the concept are identified.

The impact of risk management practice upon the implementation of recovery-oriented care in community mental health services: a qualitative investigation.

BACKGROUND: Recovery-oriented care has become guiding principle for mental health policies and practice in the UK and elsewhere. However, a pre-existing culture of risk management practice may impact upon the provision of recovery-oriented mental health services. AIMS: To explore how risk management practice impacts upon the implementation of recovery-oriented care within community mental health services. METHOD: Semi-structured interviews using vignettes were conducted with eight mental health worker and service user dyads. Grounded theory techniques were used to develop explanatory themes. RESULTS: Four themes arose: (1) recovery and positive risk taking; (2) competing frameworks of practice; (3) a hybrid of risk and recovery; (4) real-life recovery in the context of risk. DISCUSSION: In abstract responses to the vignettes, mental health workers described how they would use a positive-risk taking approach in support of recovery. In practice, this was restricted by a risk-averse culture embedded within services. Mental health workers set conditions with which service users complied to gain some responsibility for recovery. CONCLUSION: A lack of strategic guidance at policy level and lack of support and guidance at practice level may result in resistance to implementing ROC in the context of RMP. Recommendations are made for policy, training and future research.

The experiences of detained mental health service users: issues of dignity in care.

BACKGROUND: When mental health service users are detained under a Section of the Mental Health Act (MHA), they must remain in hospital for a specific time period. This is often against their will, as they are considered a danger to themselves and/or others. By virtue of being detained, service users are assumed to have lost control of an element of their behaviour and as a result their dignity could be compromised. Caring for detained service users has particular challenges for healthcare professionals. Respecting the dignity of others is a key element of the code of conduct for health professionals. Often from the service user perspective this is ignored. METHODS: This paper reports on the experiences of 19 adult service users who were, at the time of interview, detained under a Section of the MHA. These service users had experienced coercive interventions and they gave their account of how they considered their dignity to be protected (or not), and their sense of self respected (or not). RESULTS: The service users considered their dignity and respect compromised by 1) not being 'heard' by staff members, 2) a lack of involvement in decision-making regarding their care, 3) a lack of information about their treatment plans particularly medication, 4) lack of access to more talking therapies and therapeutic engagement, and 5) the physical setting/environment and lack of daily activities to alleviate their boredom. CONCLUSIONS: Dignity and respect are important values in recovery and practitioners need time to engage with service user narratives and to reflect on the ethics of their practice.

Reaching a consensus on service-user involvement in courses for professionals.

AIM: To describe how a workshop that used a modified nominal group technique (NGT) was used at the end of a research project to develop a standard of education and training requiring UK education providers to include service users in the design and delivery of education and training. BACKGROUND: Often the objective of a research project is to deliver a decision where there is insufficient objective evidence. In this particular instance a decision was sought on whether service users should be involved in the design and delivery of education and training for healthcare professionals and what this involvement might look like. One solution can be to use a formal approach to decision making. NGT is one of several approaches to decision making that seeks to achieve consensus among participants. DATA SOURCES: A modified NGT workshop was used and included students, service users and academic staff. DISCUSSION: This paper describes the workshop, its outcomes and points to consider when using such an approach. CONCLUSION: This paper outlines a modified NGT that was used in a workshop to complement other research techniques and provides practical tips on how to maximise the chances of the success of the approach. Modifications were necessary to address the particular challenges posed in this research. IMPLICATIONS FOR RESEARCH/PRACTICE: The modified NGT approach outlined in this paper could be used by nurses when addressing questions and issues related to service-user involvement in planning the design and delivery of education and training.

Understanding organisational and nursing behaviour changes associated with a therapeutic engagement improvement tool in acute mental health inpatient settings: A qualitative analysis.

Background: Enhancing the quality of therapeutic engagement between nurse and service user is related to positive impact on care, safety, and recovery outcomes. Achieving improved therapeutic engagement remains challenging in the acute mental health inpatient setting, characterised by complex social processes and contextual features that constrain behaviour change. The Therapeutic Engagement Questionnaire is an evidence-based tool co-produced with service users and nurses to improve therapeutic engagement. Objectives: The objectives of this quality improvement project were to identify the organisational and nursing behaviour changes associated with the Therapeutic Engagement Questionnaire and to understand the active behaviour change ingredients of the improvement tool and how they exert their influence. Design: A qualitative multi-site case study design in which data were collected from study site field notes and document review. Setting: Four acute mental health inpatient case study sites in England. Methods: Data referencing Therapeutic Engagement Questionnaire-linked behaviour change in project meeting field notes and documents from each study site were analysed using an inductive and deductive approach with thematic analysis. The Capability Opportunity Motivation-Behaviour model was employed as a theoretical framework. Findings: The therapeutic engagement tool had the capacity to prompt behaviour change across all three components of the behaviour change model: Capability - through nurses sharing good therapeutic engagement practice and use of statements in the questionnaire to build nurses' knowledge and skills; Opportunity - through organisational barriers being addressed and ward-level practice and culture changes; Motivation - through nurses' awareness of their influence on service user recovery, nurses' alertness to their therapeutic work, and connections between the therapeutic engagement tool and nursing core values. However, the tool did not accord with the values of some nurses, reported to be unmotivated by the recognition it gave their profession for contribution to service user recovery. In sites evidencing more prominent behaviour change, senior leader and ward-level agents of change played a valuable facilitative role. Conclusion: The therapeutic engagement tool had the potential to prompt behaviour changes at organisation and ward level and to the ways individual nurses therapeutically engage with service users, helping strengthen therapeutic engagement practice. Leadership at senior organisational and ward level was important to address contextual barriers to change. The project resulted in a conceptual framework to explain and understand the behaviour change techniques and functions linked to the therapeutic engagement tool. Longevity of the behaviour changes and their impact on service user quality of care requires future evaluation. Tweetable abstract: A therapeutic engagement tool can prompt organisational and nursing behaviour change in acute mental health inpatient settings.

Social cohesion among healthcare workers during COVID-19: Qualitative research in Indonesia, Nepal, and Vietnam.

Existing literature has portrayed numerous challenges that healthcare workers (HCWs) faced during the COVID-19 pandemic, such as heightened risks of transmission against the scarcity of protective equipment, burgeoning workload, and emotional distress, to name a few. However, most studies explored HCWs' experiences at the individual level rather than examining the collective responses. Exploring these experiences could reveal the social-cultural locality of the pandemic while identifying the system constraints in public health emergencies. As part of a mixed-method study on COVID-19 pandemic impacts, we analysed qualitative interview data with 129 HCWs and health-related staff to explore their experiences during the pandemic between 2020 and 2021 in Vietnam, Indonesia, and Nepal. Using Bahers' sociological framework, Community of Fate, we describe five themes reflecting the formation of a community of HCWs and the social cohesion underlying their efforts to survive hardship. The first three themes characterise the HCW community of fate, including (1) Recognition of extreme work-related danger, (2) physical and figurative closures where HCWs restrict themselves from the outside world, (3) chronic ordeals with overwhelming workload and responsibilities, encompassing recurrent mental health challenges. Against such extreme hardship, cohesive bonding and social resilience are reflected through two additional themes: (4) a mutual sense of moral and professional duty to protect communities, (5) the vertical and horizontal convergence among HCWs across levels and among government departments. We discuss these HCWs' challenges in relation to systemic vulnerabilities while advocating for increasing investment in public health and collaboration across government sectors to prepare for emergency situations.

A study on biomedical researchers' perspectives on public engagement in Southeast Asia.

Introduction: Public engagement is increasingly promoted in the scientific community. Although there are studies about researchers' perspectives on public engagement, these are predominantly from Global North settings and there is little data from the context of Southeast Asia. The Oxford University Clinical Research Unit (OUCRU) is a clinical and public health research programme with sites in Vietnam, Nepal and Indonesia. There is a dedicated public engagement team, and it is recognised as an important part of the research process.   Methods: Through this study we explored the views and needs of local researchers with regards to practicing public engagement. We obtained opinions of 70 researchers through an online survey with both open-ended and closed-ended questions.   Results: Most researchers perceived public engagement as improving public science literacy, rather than supporting public participation in science and research. While the participants largely see public engagement as a necessary practice, they experienced four main barriers to taking part in public engagement: time, lack of capacity, lack of support and personal perceptions. Most participants indicated they had somewhat to low confidence to communicate about science to the public. Experience, skill and knowledge, and personal preference emerged as factors that influence their perceived confidence for science communication. In our analysis, experience appeared to be the main factor contributing to researchers' high confidence.   Recommendations: We recommended to support researchers by not only providing them with training for skills and knowledge, but also with opportunities to conduct public engagement, and a range of methods to suit their personal styles of communicating. It is also evident that more support is needed to build an enabling institutional environment that gives researchers professional recognition for their engagement work. This study, while modest in its scope, has informed our approach to supporting researcher-led engagement, and may guide other institutes wishing to improve this.