Applying the Non-adoption, Abandonment, Scale-up, Spread and Sustainability (NASSS) framework to evaluate automated evidence synthesis in health behaviour change.

Automated tools to speed up the process of evidence synthesis are increasingly apparent within health behaviour research. This brief review explores the potential of the Non-adoption, Abandonment, Scale-up, Spread and Sustainability framework for supporting automated evidence synthesis in health behaviour change by applying it to the ongoing Human Behaviour-Change Project, which aims to revolutionize evidence synthesis within behaviour change intervention research. To increase the relevance of NASSS for health behaviour change, we recommend i) terminology changes ('condition' to 'behaviour' and 'patient' to 'end user') and ii) that it is used prospectively address complexities iteratively. We draw conclusions about i) the need to specify the organizations that will use the technology, ii) identifying what to do if interdependencies fail and iii) even though we have focused on automated evidence synthesis, NASSS would arguably be beneficial for technology developments in health behaviour change more generally, particularly for invention development.

A registered report survey of open research practices in psychology departments in the UK and Ireland.

Open research practices seek to enhance the transparency and reproducibility of research. While there is evidence of increased uptake in these practices, such as study preregistration and open data, facilitated by new infrastructure and policies, little research has assessed general uptake of such practices across psychology university researchers. The current study estimates psychologists' level of engagement in open research practices across universities in the United Kingdom and Ireland, while also assessing possible explanatory factors that may impact their engagement. Data were collected from 602 psychology researchers in the United Kingdom and Ireland on the extent to which they have implemented various practices (e.g., use of preprints, preregistration, open data, open materials). Here we present the summarized descriptive results, as well as considering differences between various categories of researcher (e.g., career stage, subdiscipline, methodology), and examining the relationship between researcher's practices and their self-reported capability, opportunity, and motivation (COM-B) to engage in open research practices. Results show that while there is considerable variability in engagement of open research practices, differences across career stage and subdiscipline of psychology are small by comparison. We observed consistent differences according to respondent's research methodology and based on the presence of institutional support for open research. COM-B dimensions were collectively significant predictors of engagement in open research, with automatic motivation emerging as a consistently strong predictor. We discuss these findings, outline some of the challenges experienced in this study, and offer suggestions and recommendations for future research. Estimating the prevalence of responsible research practices is important to assess sustained behaviour change in research reform, tailor educational training initiatives, and to understand potential factors that might impact engagement.

A primer for choosing, designing and evaluating registered reports for qualitative methods.

Registered reports are a publication format that involves peer reviewing studies both before and after carrying out research procedures. Although registered reports were originally developed to combat challenges in quantitative and confirmatory study designs, today registered reports are also available for qualitative and exploratory work. This article provides a brief primer that aims to help researchers in choosing, designing, and evaluating registered reports, which are driven by qualitative methods.

Teaching open and reproducible scholarship: a critical review of the evidence base for current pedagogical methods and their outcomes.

In recent years, the scientific community has called for improvements in the credibility, robustness and reproducibility of research, characterized by increased interest and promotion of open and transparent research practices. While progress has been positive, there is a lack of consideration about how this approach can be embedded into undergraduate and postgraduate research training. Specifically, a critical overview of the literature which investigates how integrating open and reproducible science may influence student outcomes is needed. In this paper, we provide the first critical review of literature surrounding the integration of open and reproducible scholarship into teaching and learning and its associated outcomes in students. Our review highlighted how embedding open and reproducible scholarship appears to be associated with (i) students' scientific literacies (i.e. students' understanding of open research, consumption of science and the development of transferable skills); (ii) student engagement (i.e. motivation and engagement with learning, collaboration and engagement in open research) and (iii) students' attitudes towards science (i.e. trust in science and confidence in research findings). However, our review also identified a need for more robust and rigorous methods within pedagogical research, including more interventional and experimental evaluations of teaching practice. We discuss implications for teaching and learning scholarship.

Opportunities, challenges and tensions: Open science through a lens of qualitative social psychology.

In recent years, there has been a focus in social psychology on efforts to improve the robustness, rigour, transparency and openness of psychological research. This has led to a plethora of new tools, practices and initiatives that each aim to combat questionable research practices and improve the credibility of social psychological scholarship. However, the majority of these efforts derive from quantitative, deductive, hypothesis-testing methodologies, and there has been a notable lack of in-depth exploration about what the tools, practices and values may mean for research that uses qualitative methodologies. Here, we introduce a Special Section of BJSP: Open Science, Qualitative Methods and Social Psychology: Possibilities and Tensions. The authors critically discuss a range of issues, including authorship, data sharing and broader research practices. Taken together, these papers urge the discipline to carefully consider the ontological, epistemological and methodological underpinnings of efforts to improve psychological science, and advocate for a critical appreciation of how mainstream open science discourse may (or may not) be compatible with the goals of qualitative research.

Back Onside protocol: A physical activity intervention to improve health outcomes in people who are unemployed or at risk of unemployment.

Given the effects of physical activity on people's mental and physical health, a better understanding is needed of how physical activity interventions may impact the health of people who are unemployed or at risk of unemployment. This has added urgency in the context of rising rates of poverty-related unemployment in the UK in 2022. The current paper details the protocol used in the evaluation of the Back Onside Programme; a community-based programme delivered by the Bradford Bulls Foundation in the Bradford District. The Programme supports people from low socio-economic backgrounds who are unemployed or at risk of unemployment to maintain regular physical activity through a ten-week physical activity intervention. This pilot study evaluates how a physical activity intervention may impact the mental and physical health of people who are unemployed or at risk of unemployment in an uncontrolled pragmatic pilot study. Four cohorts run back-to-back between May 2021 and May 2022, with separate groups for men (N = 100) and women (N = 60). Physical and wellbeing assessment at baseline and post-intervention is conducted. If the intervention works in this context for these individuals, it will be a promising low-cost community-based intervention for people who are unemployed or at risk of unemployment.

Improving evidence-based practice through preregistration of applied research: Barriers and recommendations.

Preregistration is the practice of publicly publishing plans on central components of the research process before access to, or collection, of data. Within the context of the replication crisis, open science practices like preregistration have been pivotal in facilitating greater transparency in research. However, such practices have been applied nearly exclusively to basic academic research, with rare consideration of the relevance to applied and consultancy-based research. This is particularly problematic as such research is typically reported with very low levels of transparency and accountability despite being disseminated as influential gray literature to inform practice. Evidence-based practice is best served by an appreciation of multiple sources of quality evidence, thus the current review considers the potential of preregistration to improve both the accessibility and credibility of applied research toward more rigorous evidence-based practice. The current three-part review outlines, first, the opportunities of preregistration for applied research, and second, three barriers - practical challenges, stakeholder roles, and the suitability of preregistration. Last, this review makes four recommendations to overcome these barriers and maximize the opportunities of preregistration for academics, industry, and the structures they are held within - changes to preregistration templates, new types of templates, education and training, and recognition and structural changes.

Correction: Loss of the BMP Antagonist, SMOC-1, Causes Ophthalmo-Acromelic (Waardenburg Anophthalmia) Syndrome in Humans and Mice.

[This corrects the article DOI: 10.1371/journal.pgen.1002114.].

When Your Pregnancy Echoes Your Illness: Transition to Motherhood With Inflammatory Bowel Disease.

Our aim is to provide an understanding of the experience of women with inflammatory bowel disease (IBD) who have made the transition to motherhood. A total of 22 mothers with IBD were recruited from around the United Kingdom. Semi-structured interviews were conducted and analyzed using thematic analysis. The central concept- Blurred Lines-offers a novel frame for understanding the transition to motherhood with IBD through identifying parallels between having IBD and becoming, and being, a mother. Parallels clustered into three main themes: Need for Readiness, Lifestyle Changes, and Monitoring Personal and Physical Development. Hence, women with IBD are in some ways well prepared for the challenges of motherhood even though, as a group, they tend to restrict their reproductive choices. We recommend health professionals initiate conversations about reproduction early and provide a multidisciplinary approach to pregnancy and IBD in which women have confidence that their ongoing treatment will be integrated successfully with their maternity care.

Rewarding altruism: addressing the issue of payments for volunteers in public health initiatives.

Lay involvement in public health programmes occurs through formalised lay health worker (LHW) and other volunteer roles. Whether such participation should be supported, or indeed rewarded, by payment is a critical question. With reference to policy in England, UK, this paper argues how framing citizen involvement in health only as time freely given does not account for the complexities of practice, nor intrinsic motivations. The paper reports results on payment drawn from a study of approaches to support lay people in public health roles, conducted in England, 2007-9. The first phase of the study comprised a scoping review of 224 publications, three public hearings and a register of projects. Findings revealed the diversity of approaches to payment, but also the contested nature of the topic. The second phase investigated programme support matters in five case studies of public health projects, which were selected primarily to reflect role types. All five projects involved volunteers, with two utilising forms of payment to support engagement. Interviews were conducted with a sample of project staff, LHWs (paid and unpaid), external partners and service users. Drawing on both lay and professional perspectives, the paper explores how payment relates to social context as well as various motivations for giving, receiving or declining financial support. The findings show that personal costs are not always absorbed, and that there is a potential conflict between financial support, whether sessional payment or expenses, and welfare benefits. In identifying some of the advantages and disadvantages of payment, the paper highlights the complexity of an issue often addressed only superficially. It concludes that, in order to support citizen involvement, fairness and value should be considered alongside pragmatic matters of programme management; however policy conflicts need to be resolved to ensure that employment and welfare rights are maintained.

Masculinities, humour and care for penile cancer: a qualitative study.

AIM: To explore how men with penile cancer construct humour in relation to their diagnosis and treatment. BACKGROUND: Functionalist, relief and incongruity theories attempt to account for humour, but there is a dearth of empirical evidence in nursing care. This is particularly so in relation to a condition like penile cancer where some nurses think that humour in their interactions with patients would be inappropriate. DESIGN: The study employed a participative, mixed-qualitative-methods design. METHOD: Focus groups and patient-conducted interviews were both used during a one-day 'pilot workshop' in March 2011. The data were initially analysed using framework analysis. This paper explores the theme of humour in depth. FINDINGS: Humour helped participants make light of their condition, which meant that they could laugh about the consequences of treatment ('laughing about urination') and build rapport with health professionals ('humour with health professionals'). Nevertheless, the use of humour was less important than the treatment of their cancer ('humour discounted') and there was a fear that they would be subject to ridicule because of their condition ('fear of ridicule'). CONCLUSION: The findings suggest a combination of functionalist, relief and incongruity theories of humour; the emotions these men experience are contained (functionalist) and released (relief) through humorous interaction, and the potential for comedy lies in an incongruity between what is expected socially and the experiences of these men, for example, around expectations that men use urinals in public toilets. Nurses should continue to use humour to build rapport with patients, should they judge this to be appropriate, although they may want to avoid jokes about sexual and urinary functioning until after treatment.

Engaging men with penile cancer in qualitative research: reflections from an interview-based study.

AIM: To explore the challenges of engaging men with penile cancer in qualitative interview research. BACKGROUND: Qualitative interviewing offers an ideal tool for exploring men's experiences of illness, complementing and providing context to gendered health inequalities identified in epidemiological research on men. But conducting interviews with men can be challenging and embarking on a qualitative interview study with males can feel like a daunting task, given the limited amount of practical, gender-sensitive guidance for researchers. Reflecting on a researcher's experience of conducting qualitative research on men with penile cancer, this paper explores the potential challenges of interviewing this group, but also documents how engagement and data collection were achieved. REVIEW METHODS: This is a reflective paper, informed by the experiences of a male researcher (KW) with no nurse training, who conducted 28 interviews with men who had been treated for penile cancer. The researcher's experiences are reported in chronological order, from the methodological challenges of recruitment to those of conducting the interview. IMPLICATIONS FOR PRACTICE/RESEARCH: The paper offers a resource for the novice researcher, highlighting some advantages and disadvantages of conducting qualitative interview research as a nurse researcher, as well as recommendations on how to overcome challenges. CONCLUSION: Engaging men with penile cancer in qualitative interview raises practical, methodological, ethical and emotional challenges for the researcher. However, when these challenges are met, men will talk about their health. Methodological procedures must enable an open and ongoing dialogue with clinical gatekeepers and potential participants to promote engagement. Support from colleagues is essential for any interviewer, no matter how experienced the researcher is.

Championing mental health at work: emerging practice from innovative projects in the UK.

This paper examines the value of participatory approaches within interventions aimed at promoting mental health and wellbeing in the workplace. Specifically the paper explores data from the thematic evaluation of the Mental Health and Employment project strand within the Altogether Better programme being implemented in England in the Yorkshire and Humber region, which was funded through the BIG Lottery and aimed to empower people across the region to lead better lives. The evaluation combined a systematic evidence review with semi-structured interviews across mental health and employment projects. Drawing on both evaluation elements, the paper examines the potential of workplace-based 'business champions' to facilitate organizational culture change within enterprises within a deprived regional socio-economic environment. First, the paper identifies key policy drivers for interventions around mental health and employment, summarizes evidence review findings and describes the range of activities within three projects. The role of the 'business champion' emerged as crucial to these interventions and therefore, secondly, the paper examines how champions' potential to make a difference depends on the work settings and their existing roles, skills and motivation. In particular, champions can proactively coordinate project strands, embed the project, encourage participation, raise awareness, encourage changes to work procedures and strengthen networks and partnerships. The paper explores how these processes can facilitate changes in organizational culture. Challenges of implementation are identified, including achieving leverage with senior management, handover of ownership to fellow employees, assessing impact and sustainability. Finally, implications for policy and practice are discussed, and conclusions drawn concerning the roles of champions within different workplace environments.

The impact of surgical treatment for penile cancer -- patients' perspectives.

PURPOSE OF THE RESEARCH: Penile cancer is a rare but highly treatable condition. Whilst over 80% survive for over five years, treatment can have a significant impact on quality of life. There has been little research conducted to date on men's experiences of treatment for penile cancer. The Patients Experiences of Penile Cancer study (PEPC) aimed to redress this shortfall by exploring men's experiences of surgical treatment for penile cancer. METHODS AND SAMPLE: The study used a narrative history design in which data were collected using one-on-one semi-structured interviews. Maximum variation sampling was used to acquire the widest possible range of experiences. Twenty-seven interviews of around one hour were conducted with men with an average age of 63 years at diagnosis (range = 41-82). The data were analysed using constant comparison analysis. KEY RESULTS: The physical impact of surgery was inter-connected with broader events in the lives of the men experiencing treatment. These experiences cover urinary function, sexual function and sexual relationships, healing and recovery, masculinity, mental well-being, coping and support. CONCLUSION: A key area for the development of care is to devise and evaluate procedures for ensuring that men are well-informed about the extent and potential consequences of their treatment. Men's experiences of penile cancer surgery will be informed by a complex web interlaced with their broader lives, making it difficult for health professionals to judge how surgery will impact on a men presenting to them. Further research is required to ascertain the most appropriate strategies for rehabilitation of men experiencing penile cancer surgery.

Miller (Genee-Wiedemann) syndrome represents a clinically and biochemically distinct subgroup of postaxial acrofacial dysostosis associated with partial deficiency of DHODH.

Biallelic mutations in the gene encoding DHOdehase [dihydroorotate dehydrogenase (DHODH)], an enzyme required for de novo pyrimidine biosynthesis, have been identified as the cause of Miller (Genée-Weidemann or postaxial acrofacial dysostosis) syndrome (MIM 263750). We report compound heterozygous DHODH mutations in four additional families with typical Miller syndrome. Complementation in auxotrophic yeast demonstrated reduced pyrimidine synthesis and in vitro enzymatic analysis confirmed reduced DHOdehase activity in 11 disease-associated missense mutations, with 7 alleles showing discrepant activity between the assays. These discrepancies are partly explained by the domain structure of DHODH and suggest both assays are useful for interpretation of individual alleles. However, in all affected individuals, the genotype predicts that there should be significant residual DHOdehase activity. Urine samples obtained from two mutation-positive cases showed elevated levels of orotic acid (OA) but not dihydroorotate (DHO), an unexpected finding since these represent the product and the substrate of DHODH enzymatic activity, respectively. Screening of four unrelated cases with overlapping but atypical clinical features showed no mutations in either DHODH or the other de novo pyrimidine biosynthesis genes (CAD, UMPS), with these cases also showing normal levels of urinary OA and DHO. In situ analysis of mouse embryos showed Dhodh, Cad and Umps to be strongly expressed in the pharyngeal arch and limb bud, supporting a site- and stage-specific requirement for de novo pyrimidine synthesis. The developmental sensitivity to reduced pyrimidine synthesis capacity may reflect the requirement for an exceptional mitogenic response to growth factor signalling in the affected tissues.

'Straight to the GP; that would be where I would go': an analysis of male frequent attenders' constructions of their decisions to use or not use health-care services in the UK.

An important consideration that needs adding to discussions of patient choice and whether or not men are reluctant to use primary care services is that many frequent attenders are male. The aim of this article is to explore how male frequent attenders construct decisions to use or not use health-care services. This is achieved through secondary analysis of baseline interviews with male frequent attenders from the Self Care in Primary Care study. As this was a complex study, a three-step analytic process was employed to incorporate the involvement of multiple researchers working together over a number of years. First, the interviewer summarised each interview and second, the summaries were read as a group to find themes across them. Subsequently, we returned to the interviews to add detail that would further illustrate or challenge the analysis. Participants talked of 'engaging health and avoiding ill-health', constructing themselves as embodied, health conscious and rational in a similar vein to constructions of feminine interactions with health. While participants talked of 'choosing health services' as if they were drawing upon a range of choices, the dominance of the image of the GP was such that seeing a GP was the only legitimate health choice.

'Think differently and be prepared to demonstrate trust': findings from public hearings, England, on supporting lay people in public health roles.

Professional support processes are critical for the establishment and maintenance of community health worker programmes. This paper reports on three public hearings held in England, UK, that were conducted as part of a national study into approaches to develop and support lay people in public health roles. Individuals with relevant theoretical or practical expertise, including lay activists, presented evidence in public as expert witnesses. Formal presentations, questions and plenary discussions were recorded and later analysed as qualitative data. This paper presents the results and critically examines emergent issues relating to the sustainability of lay health worker programmes. Consideration is given to the diversity of contemporary practice in England. Barriers seen to affect sustainability included organizational culture and onerous bureaucratic processes. Major themes emerging from the expert evidence included recruitment and training strategies, financial support and the need for a robust infrastructure. The expert hearings, in creating a public space for deliberation, opened up discussion on the levels and type of programme support required to foster lay health worker programmes. The paper concludes that professional support needs to be accompanied by a reorientation of public services to support lay engagement in programme delivery.

Loss of the BMP antagonist, SMOC-1, causes Ophthalmo-acromelic (Waardenburg Anophthalmia) syndrome in humans and mice.

Ophthalmo-acromelic syndrome (OAS), also known as Waardenburg Anophthalmia syndrome, is defined by the combination of eye malformations, most commonly bilateral anophthalmia, with post-axial oligosyndactyly. Homozygosity mapping and subsequent targeted mutation analysis of a locus on 14q24.2 identified homozygous mutations in SMOC1 (SPARC-related modular calcium binding 1) in eight unrelated families. Four of these mutations are nonsense, two frame-shift, and two missense. The missense mutations are both in the second Thyroglobulin Type-1 (Tg1) domain of the protein. The orthologous gene in the mouse, Smoc1, shows site- and stage-specific expression during eye, limb, craniofacial, and somite development. We also report a targeted pre-conditional gene-trap mutation of Smoc1 (Smoc1(tm1a)) that reduces mRNA to ∼10% of wild-type levels. This gene-trap results in highly penetrant hindlimb post-axial oligosyndactyly in homozygous mutant animals (Smoc1(tm1a/tm1a)). Eye malformations, most commonly coloboma, and cleft palate occur in a significant proportion of Smoc1(tm1a/tm1a) embryos and pups. Thus partial loss of Smoc-1 results in a convincing phenocopy of the human disease. SMOC-1 is one of the two mammalian paralogs of Drosophila Pentagone, an inhibitor of decapentaplegic. The orthologous gene in Xenopus laevis, Smoc-1, also functions as a Bone Morphogenic Protein (BMP) antagonist in early embryogenesis. Loss of BMP antagonism during mammalian development provides a plausible explanation for both the limb and eye phenotype in humans and mice.

Gain-of-function mutations of ARHGAP31, a Cdc42/Rac1 GTPase regulator, cause syndromic cutis aplasia and limb anomalies.

Regulation of cell proliferation and motility is essential for normal development. The Rho family of GTPases plays a critical role in the control of cell polarity and migration by effecting the cytoskeleton, membrane trafficking, and cell adhesion. We investigated a recognized developmental disorder, Adams-Oliver syndrome (AOS), characterized by the combination of aplasia cutis congenita (ACC) and terminal transverse limb defects (TTLD). Through a genome-wide linkage analysis, we detected a locus for autosomal-dominant ACC-TTLD on 3q generating a maximum LOD score of 4.93 at marker rs1464311. Candidate-gene- and exome-based sequencing led to the identification of independent premature truncating mutations in the terminal exon of the Rho GTPase-activating protein 31 gene, ARHGAP31, which encodes a Cdc42/Rac1 regulatory protein. Mutant transcripts are stable and increase ARHGAP31 activity in vitro through a gain-of-function mechanism. Constitutively active ARHGAP31 mutations result in a loss of available active Cdc42 and consequently disrupt actin cytoskeletal structures. Arhgap31 expression in the mouse is substantially restricted to the terminal limb buds and craniofacial processes during early development; these locations closely mirror the sites of impaired organogenesis that characterize this syndrome. These data identify the requirement for regulated Cdc42 and/or Rac1 signaling processes during early human development.