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Additional file 1: GRIPP2 reporting checklists: tools to improve reporting of patient and public involvement in research.

Additional file 1. GRIPP2 Short Form Checklist.

Supplemental material, sj-docx-1-jvi-10.1177_02646196221112800 for Impact of Charles Bonnet Syndrome on visually impaired older adults’ ability to engage in physical activity: A scoping review by Katharine Fisher, Caroline Sanders and Emma Stanmore in The British Journal of Visual Impairment

IntroductionThe early years (ages 0-8) is a critical stage of childhood development, supporting families during this time requires collective efforts across sectors. Siloed streams limit our ability to meet families’ needs thereby missing opportunities to positively influence childhood outcomes. In northern British Columbia a small group of professionals completed training in Compassionate Systems Leadership (CSL). The CSL approach focuses on developing shared knowledge and cultural awareness to strengthen capacity to effectively progress systems change initiatives. Applying CSL with a wider community across northern BC allowed the team to examine how all partners could address silos in early years services. Aims Objectives Theory or MethodsA first step in applying CSL is to be culturally sensitive and actively relational with professionals working within early years services. In February 2021 we hosted a summit in northern BC, bringing together professionals across child and family serving sectors. The objectives were to find common ground, celebrate local work supporting children, and build on understanding priorities in the early years. Our five day event wove together Indigenous and western pedagogies, highlighting northern First Nation voices and cultural practices. The event used a hybrid model of pre-recorded asynchronous sessions with a concluding full day virtual workshop. Highlights or Results or Key FindingsEarly years services were described as patchy and inconsistent. Critically, the burden of weaving together supports falls to primary caregivers, and across sectors it was acknowledged this created gaps in comprehensive early years services. Many communities worked hard to lessen fragmentation and participants described how relational ways of being helped co-create partnered supports. Participants acknowledged that working compassionately facilitated local initiatives which positively impacted the early years, yet gaps remained including the absence of health’s engagement in work across the region. There was consensus that a holistic, culturally respectful, multidisciplinary team, which includes primary health care was needed to support the health and development needs of young children. Discussion focused on expanding hub models of care which exist in select northern communities. A clear message from the summit was a willingness for cross-sector collaboration, and the development of an integrated approach to support families in the early years is required.ConclusionsIn facilitating a safe space that allowed for vulnerability and relational ways of engaging across sectors we discovered commitment, interest, and a willingness for those present to consider new ideas and partnerships that would allow for greater integration of early years services in northern BC’s rural contexts. Implications for applicability/transferability sustainability and limitations Shifting from siloed work to integration across services is complex. Relational ways of being helped providers with this complexity, creating space to learn from each other, begin co-creation, and enhance partnerships. This was evident in early years services but is likely to apply in other service areas seeking integration.

Additional file 3. The DEPEND study toolkit.

Additional file 1. GRIPP2 long form.

Additional file 4. A new approach to collecting verbal feedback.

Additional file 2. Example trigger discussion slides used in the co-design process.

Purpose Developing technological innovations in healthcare is made complex and difficult due to effects upon the practices of professional, managerial and other stakeholders. Drawing upon the concept of boundary object, this paper explores the challenges of achieving effective collaboration in the development and use of a novel healthcare innovation in the English healthcare system. Design/methodology/approach A case study is presented of the development and implementation of a smart phone application (app) for use by rheumatoid arthritis patients. Over a two-year period (2015–2017), qualitative data from recorded clinical consultations (n = 17), semi-structured interviews (n = 63) and two focus groups (n = 13) were obtained from participants involved in the app's development and use (clinicians, patients, researchers, practitioners, IT specialists and managers). Findings The case focuses on the use of the app and its outputs as a system of inter-connected boundary objects. The analysis highlights the challenges overcome in the innovation's development and how knowledge sharing between patients and clinicians was enhanced, altering the nature of the clinical consultation. It also shows how conditions surrounding the innovation both enabled its development and inhibited its wider scale-up. Originality/value By recognizing that technological artefacts can simultaneously enable and inhibit collaboration, this paper highlights the need to overcome tensions between the transformative capability of such healthcare innovations and the inhibiting effects simultaneously created on change at a wider system level.

Additional file 1. Search Strategy

Plain English summary There are a growing number of mobile phones, watches and electronic devices which can be worn on the body to track aspects of health and well-being, such as daily steps, sleep and exercise. Dementia researchers think that these devices could potentially be used as part of future research projects, for example to help spot changes in daily activity that may signal the early symptoms of dementia. We asked a range of older people, including people living with dementia and their carers, to participate in interactive discussions about how future participants might find using these devices as part of research projects. We also invited volunteers to borrow a range of devices to test at home, giving them further insights. Discussions revealed that people were generally supportive of this type of research, provided they gave informed consent and that devices were discreet, comfortable and easy to use. They also valued technical support and regular feedback on study progress to encourage ongoing participation. These findings were used to develop a pool of devices for researchers, with computer software and written guidance to help plan, design and support studies. Our work shows that when given the right opportunities, people who are affected by dementia can provide valuable insights that can enhance the design, delivery and quality of future research. Abstract Background Increasingly, researchers are recognising the potential for connected health devices, including smartphones and smartwatches, to generate high resolution data about patterns of daily activity and health outcomes. One aim of the Dementias Platform UK (DPUK) project is to provide researchers with a secure means to collect, collate and link data generated by such devices, thereby accelerating this type of research in the field of dementia. We aimed to involve members of the public in discussions about the acceptability and feasibility of different devices and research designs to inform the development of a device pool, software platform and written guidance to support future studies. Methods Over 30 people attended a series of interactive workshops, drop-in sessions and meetings in Greater Manchester. This included people living with dementia and cognitive impairments, carers and people without memory problems. Discussions were tailored to suit different audiences and focused on the feasibility and acceptability of a range of different wearable devices and research designs. We also invited volunteers to borrow a device to test at home, enabling further insights from hands-on interactions with devices. Results Discussions revealed that people were supportive of connected health dementia research in principle, provided they gave informed consent and that devices were discreet, comfortable and easy to use. Moreover, they recommended technical support and regular feedback on study progress to encourage ongoing participation. Conclusion By using a range of discussion-based and practical activities, we found it was feasible to involve people affected by dementia and use their insights to shape the development of a software platform and device pool to support future connected health dementia research. We recommend that researchers planning such studies in future pay adequate attention to designing suitable participant information, technical support and mechanisms of providing study progress updates to support sustained engagement from participants.

ObjectivesTo establish the acceptability and feasibility of collecting daily patient-generated health data (PGHD) using smartphones and integrating PGHD into the electronic health record (EHR), using the example of rheumatoid arthritis (RA). MethodsThe Remote Monitoring in RA (REMORA) smartphone app was co-designed with patients, clinicians and researchers using qualitative semi-structured interviews and focus groups, including selection of question sets for symptoms and disease impact. PGHD were integrated into the EHR of one hospital and available in graphical form during consultations. Acceptability and feasibility were assessed with 20 RA patients and two clinicians over three months. A qualitative evaluation included semi-structured interviews with patients and clinicians before and after using the app, and audio-recordings of consultations to explore impact on the consultation. PGHD completeness was summarised descriptively, and qualitative data were analysed thematically. ResultsPatients submitted data on a median of 91% days over three months. Qualitative analysis generated three themes: RA as an invisible disease; providing the bigger picture of RA; and enabling person-centred consultations. Themes demonstrated the system helped render patients’ RA more visible by providing the “bigger picture”, identifying real-time changes in disease activity and capturing symptoms that would otherwise have been missed. Graphical summaries during consultations enabled a more person-centred approach whereby patients felt better able to participate in consultations and treatment plans.ConclusionREMORA has uniquely integrated daily PGHD from smartphones into the EHR. It has delivered proof-of-concept that such integrated remote monitoring systems are feasible and can transform consultations for clinician and patient benefit.

BACKGROUND: The Knee OsteoArthritis, Linking Activity and Pain (KOALAP) study is the first to test the feasibility of using consumer-grade cellular smartwatches for health care research. OBJECTIVE: The overall aim was to investigate the feasibility of using consumer-grade cellular smartwatches as a novel tool to capture data on pain (multiple times a day) and physical activity (continuously) in patients with knee osteoarthritis. Additionally, KOALAP aimed to investigate smartwatch sensor data quality and assess whether engagement, acceptability, and user experience are sufficient for future large-scale observational and interventional studies. METHODS: A total of 26 participants with self-diagnosed knee osteoarthritis were recruited in September 2017. All participants were aged 50 years or over and either lived in or were willing to travel to the Greater Manchester area. Participants received a smartwatch (Huawei Watch 2) with a bespoke app that collected patient-reported outcomes via questionnaires and continuous watch sensor data. All data were collected daily for 90 days. Additional data were collected through interviews (at baseline and follow-up) and baseline and end-of-study questionnaires. This study underwent full review by the University of Manchester Research Ethics Committee (#0165) and University Information Governance (#IGRR000060). For qualitative data analysis, a system-level security policy was developed in collaboration with the University Information Governance Office. Additionally, the project underwent an internal review process at Google, including separate reviews of accessibility, product engineering, privacy, security, legal, and protection regulation compliance. RESULTS: Participants were recruited in September 2017. Data collection via the watches was completed in January 2018. Collection of qualitative data through patient interviews is still ongoing. Data analysis will commence when all data are collected; results are expected in 2019. CONCLUSIONS: KOALAP is the first health study to use consumer cellular smartwatches to collect self-reported symptoms alongside sensor data for musculoskeletal disorders. The results of this study will be used to inform the design of future mobile health studies. Results for feasibility and participant motivations will inform future researchers whether or under which conditions cellular smartwatches are a useful tool to collect patient-reported outcomes alongside passively measured patient behavior. The exploration of associations between self-reported symptoms at different moments will contribute to our understanding of whether it may be valuable to collect symptom data more frequently. Sensor data-quality measurements will indicate whether cellular smartwatch usage is feasible for obtaining sensor data. Methods for data-quality assessment and data-processing methods may be reusable, although generalizability to other clinical areas should be further investigated. INTERNATIONAL REGISTERED REPORT IDENTIFIER (IRRID): DERR1-10.2196/10238.

Background:The huge increase in smartphone use heralds an enormous opportunity for epidemiology research, but there is limited evidence regarding long-term engagement and attrition in mobile health (mHealth) studies.Objective:The objective of this study was to examine how representative the Cloudy with a Chance of Pain study population is of wider chronic-pain populations and to explore patterns of engagement among participants during the first 6 months of the study.Methods:Participants in the United Kingdom who had chronic pain (≥3 months) and enrolled between January 20, 2016 and January 29, 2016 were eligible if they were aged ≥17 years and used the study app to report any of 10 pain-related symptoms during the study period. Participant characteristics were compared with data from the Health Survey for England (HSE) 2011. Distinct clusters of engagement over time were determined using first-order hidden Markov models, and participant characteristics were compared between the clusters.Results:Compared with the data from the HSE, our sample comprised a higher proportion of women (80.51%, 5129/6370 vs 55.61%, 4782/8599) and fewer persons at the extremes of age (16-34 and 75+). Four clusters of engagement were identified: high (13.60%, 865/6370), moderate (21.76%, 1384/6370), low (39.35%, 2503/6370), and tourists (25.44%, 1618/6370), between which median days of data entry ranged from 1 (interquartile range; IQR: 1-1; tourist) to 149 (124-163; high). Those in the high-engagement cluster were typically older, whereas those in the tourist cluster were mostly male. Few other differences distinguished the clusters.Conclusions:Cloudy with a Chance of Pain demonstrates a rapid and successful recruitment of a large, representative, and engaged sample of people with chronic pain and provides strong evidence to suggest that smartphones could provide a viable alternative to traditional data collection methods.

Background: The increasing ownership of smartphones provides major opportunities for epidemiological research through self-reported and passively collected data.Objective: This pilot study aimed to codesign a smartphone app to assess associations between weather and joint pain in patients with rheumatoid arthritis (RA) and to study the success of daily self-reported data entry over a 60-day period and the enablers of and barriers to data collection.Methods: A patient and public involvement group (n=5) and 2 focus groups of patients with RA (n=9) supported the codesign of the app collecting self-reported symptoms. A separate “capture app” was designed to collect global positioning system (GPS) and continuous raw accelerometer data, with the GPS-linking providing local weather data. A total of 20 patients with RA were then recruited to collect daily data for 60 days, with entry and exit interviews. Of these, 17 were loaned an Android smartphone, whereas 3 used their own Android smartphones.Results: Of the 20 patients, 6 (30%) withdrew from the study: 4 because of technical challenges and 2 for health reasons. The mean completion of daily entries was 68% over 2 months. Patients entered data at least five times per week 65% of the time. Reasons for successful engagement included a simple graphical user interface, automated reminders, visualization of data, and eagerness to contribute to this easily understood research question. The main barrier to continuing engagement was impaired battery life due to the accelerometer data capture app. For some, successful engagement required ongoing support in using the smartphones.Conclusions: This successful pilot study has demonstrated that daily data collection using smartphones for health research is feasible and achievable with high levels of ongoing engagement over 2 months. This result opens important opportunities for large-scale longitudinal epidemiological research.

BackgroundThe increasing ownership of smartphones provides major opportunities for epidemiological research through self-reported and passively collected data. ObjectiveThis pilot study aimed to codesign a smartphone app to assess associations between weather and joint pain in patients with rheumatoid arthritis (RA) and to study the success of daily self-reported data entry over a 60-day period and the enablers of and barriers to data collection. MethodsA patient and public involvement group (n=5) and 2 focus groups of patients with RA (n=9) supported the codesign of the app collecting self-reported symptoms. A separate “capture app” was designed to collect global positioning system (GPS) and continuous raw accelerometer data, with the GPS-linking providing local weather data. A total of 20 patients with RA were then recruited to collect daily data for 60 days, with entry and exit interviews. Of these, 17 were loaned an Android smartphone, whereas 3 used their own Android smartphones. ResultsOf the 20 patients, 6 (30%) withdrew from the study: 4 because of technical challenges and 2 for health reasons. The mean completion of daily entries was 68% over 2 months. Patients entered data at least five times per week 65% of the time. Reasons for successful engagement included a simple graphical user interface, automated reminders, visualization of data, and eagerness to contribute to this easily understood research question. The main barrier to continuing engagement was impaired battery life due to the accelerometer data capture app. For some, successful engagement required ongoing support in using the smartphones. ConclusionsThis successful pilot study has demonstrated that daily data collection using smartphones for health research is feasible and achievable with high levels of ongoing engagement over 2 months. This result opens important opportunities for large-scale longitudinal epidemiological research.

Data coding tree (DOCX 22 kb)

COnsolidated criteria for REporting Qualitative research (COREQ) Checklist (DOCX 27 kb)

MAXimising Involvement in MUltiMorbidity (MAXIMUM) in Primary Care - Patient participant medical information form (DOCX 50 kb)

Background: The ethics of research into online communities is a long-debated issue, with many researchers arguing that open-access discussion groups are publically accessible data and do not require informed consent from participants for their use for research purposes. However, it has been suggested that there is a discrepancy between the perceived and actual privacy of user-generated online content by community members.Objective: There has been very little research regarding how privacy is experienced and enacted online. The objective of this study is to address this gap by qualitatively exploring the expectations of privacy on Internet forums among individuals with long-term conditions.Methods: Semistructured interviews were conducted with 20 participants with myalgic encephalomyelitis/chronic fatigue syndrome (ME/CFS) and 21 participants with type 1 and 2 diabetes mellitus, and were analyzed using thematic analysis. Participants were recruited via online and offline routes, namely forums, email lists, newsletters, and face-to-face support groups.Results: The findings indicate that privacy online is a nebulous concept. Rather than individuals drawing a clear-cut distinction between what they would and would not be comfortable sharing online, it was evident that these situations were contextually dependent and related to a number of unique and individual factors.Conclusions: Interviewees were seen to carefully manage how they presented themselves on forums, filtering and selecting the information that they shared about themselves in order to develop and maintain a particular online persona, while maintaining and preserving an acceptable level of privacy.

Background The ethics of research into online communities is a long-debated issue, with many researchers arguing that open-access discussion groups are publically accessible data and do not require informed consent from participants for their use for research purposes. However, it has been suggested that there is a discrepancy between the perceived and actual privacy of user-generated online content by community members. Objective There has been very little research regarding how privacy is experienced and enacted online. The objective of this study is to address this gap by qualitatively exploring the expectations of privacy on Internet forums among individuals with long-term conditions. Methods Semistructured interviews were conducted with 20 participants with myalgic encephalomyelitis/chronic fatigue syndrome (ME/CFS) and 21 participants with type 1 and 2 diabetes mellitus, and were analyzed using thematic analysis. Participants were recruited via online and offline routes, namely forums, email lists, newsletters, and face-to-face support groups. Results The findings indicate that privacy online is a nebulous concept. Rather than individuals drawing a clear-cut distinction between what they would and would not be comfortable sharing online, it was evident that these situations were contextually dependent and related to a number of unique and individual factors. Conclusions Interviewees were seen to carefully manage how they presented themselves on forums, filtering and selecting the information that they shared about themselves in order to develop and maintain a particular online persona, while maintaining and preserving an acceptable level of privacy.

The acquisition of iron is crucial for bacterial pathogenesis. In this thesis I describe the development and validation of a biosensor for heme in bacteria, an important host source of iron for human pathogens. I demonstrate that the co-expression of a phytochrome-based fluorophore (PBF), which utilises the heme-derived metabolite biliverdin IXα as a chromophore, with a heme oxygenase (HO) in Gram-negative bacteria results in heme-dependent near infra-red fluorescence. Using this PBF+HO reporter, I probe heme metabolism in the opportunistic human pathogen Pseudomonas aeruginosa and for the first time provide direct evidence linking the previously putative heme receptor HxuC to heme metabolism. Further, coupling of the heme biosensor to transposon mutagenesis and Fluorescence-Activated Cell Sorting was used to screen a P. aeruginosa transposon library leading to the identification of several putative novel components of heme metabolism. Finally, I demonstrate the potential of our fluorescent biosensor to be used to study heme metabolism and host-pathogen interactions in vivo using a live adult zebrafish model of infection. The PBF+HO heme biosensor allows the highthroughput, non-invasive study of bacterial heme metabolism in real-time and proves a useful tool for gaining further insight into iron-heme metabolism in bacterial pathogens. Open Access

Links between presented themes and NPT components. (DOCX 16 kb)

Interview Schedule. (DOCX 17 kb)

OBJECTIVE: Computerised therapies play an integral role in efforts to improve access to psychological treatment for patients with depression and anxiety. However, despite recognised problems with uptake, there has been a lack of investigation into the barriers and facilitators of engagement. We aimed to systematically review and synthesise findings from qualitative studies of computerised therapies, in order to identify factors impacting on engagement. METHOD: Systematic review and meta-synthesis of qualitative studies of user experiences of computer delivered therapy for depression and/or anxiety. RESULTS: 8 studies were included in the review. All except one were of desktop based cognitive behavioural treatments. Black and minority ethnic and older participants were underrepresented, and only one study addressed users with a co-morbid physical health problem. Through synthesis, we identified two key overarching concepts, regarding the need for treatments to be sensitive to the individual, and the dialectal nature of user experience, with different degrees of support and anonymity experienced as both positive and negative. We propose that these factors can be conceptually understood as the 'non-specific' or 'common' factors of computerised therapy, analogous to but distinct from the common factors of traditional face-to-face therapies. CONCLUSION: Experience of computerised therapy could be improved through personalisation and sensitisation of content to individual users, recognising the need for users to experience a sense of 'self' in the treatment which is currently absent. Exploiting the common factors of computerised therapy, through enhancing perceived connection and collaboration, could offer a way of reconciling tensions due to the dialectal nature of user experience. Future research should explore whether the findings are generalisable to other patient groups, to other delivery formats (such as mobile technology) and other treatment modalities beyond cognitive behaviour therapy. The proposed model could aid the development of enhancements to current packages to improve uptake and support engagement.

Objective: To assess the effect of second generation, home based telehealth on health related quality of life, anxiety, and depressive symptoms over 12 months in patients with long term conditions.\ud \ud Design: A study of patient reported outcomes (the Whole Systems Demonstrator telehealth questionnaire study; baseline n=1573) was nested in a pragmatic, cluster randomised trial of telehealth (the Whole Systems Demonstrator telehealth trial, n=3230). General practice was the unit of randomisation, and telehealth was compared with usual care. Data were collected at baseline, four months (short term), and 12 months (long term). Primary intention to treat analyses tested treatment effectiveness; multilevel models controlled for clustering by general practice and a range of covariates. Analyses were conducted for 759 participants who completed questionnaire measures at all three time points (complete case cohort) and 1201 who completed the baseline assessment plus at least one other assessment (available case cohort). Secondary per protocol analyses tested treatment efficacy and included 633 and 1108 participants in the complete case and available case cohorts, respectively.\ud \ud Setting: Provision of primary and secondary care via general practices, specialist nurses, and hospital clinics in three diverse regions of England (Cornwall, Kent, and Newham), with established integrated health and social care systems.\ud \ud Participants: Patients with chronic obstructive pulmonary disease (COPD), diabetes, or heart failure recruited between May 2008 and December 2009.\ud \ud Main outcome measures: Generic, health related quality of life (assessed by physical and mental health component scores of the SF-12, and the EQ-5D), anxiety (assessed by the six item Brief State-Trait Anxiety Inventory), and depressive symptoms (assessed by the 10 item Centre for Epidemiological Studies Depression Scale).\ud \ud Results: In the intention to treat analyses, differences between treatment groups were small and non-significant for all outcomes in the complete case (0.480≤P≤0.904) or available case (0.181≤P≤0.905) cohorts. The magnitude of differences between trial arms did not reach the trial defined, minimal clinically important difference (0.3 standardised mean difference) for any outcome in either cohort at four or 12 months. Per protocol analyses replicated the primary analyses; the main effect of trial arm (telehealth v usual care) was non-significant for any outcome (complete case cohort 0.273≤P≤0.761; available case cohort 0.145≤P≤0.696).\ud \ud Conclusions: Second generation, home based telehealth as implemented in the Whole Systems Demonstrator Evaluation was not effective or efficacious compared with usual care only. Telehealth did not improve quality of life or psychological outcomes for patients with chronic obstructive pulmonary disease, diabetes, or heart failure over 12 months. The findings suggest that concerns about potentially deleterious effect of telehealth are unfounded for most patients.\ud \ud Trial Registration: ISRCTN43002091.

Self-care has the potential to make a significant contribution to vascular conditions, but engagement with self-care support has been limited. Lack of relevant information is highlighted by patients and policy-makers as an important barrier to effective self-care, and information provides a potentially efficient platform for changing behaviour. However, work within the social sciences has generally seen information as a necessary but insufficient driver of health behaviours. Furthermore, some groups (such as the socially disadvantaged) are expected to be less amenable to information interventions. We conducted an integrated conceptual and empirical review on information-based interventions for people with vascular disease (diabetes, heart disease and kidney disease). We reviewed conceptual and empirical work concerning the role and impact of information in self-care support to generate an explanatory framework to determine why information was effective or ineffective in encouraging self-care in patients with vascular conditions. This involved mapping relevant theories and models linking information and self-care. We also explored published systematic reviews of educational interventions in diabetes, coronary heart disease and chronic kidney disease to examine the role of information and evidence concerning its effectiveness and impact in different patient populations. The conceptual review identified variation among information interventions in terms of type, function, and their relationship to behaviour change techniques and psychological mediators of behaviour change. Key moderators of the effect of information included types of disorder, and patient capacity and resources. A wealth of educational interventions exists for diabetes and heart conditions, but the precise components of these interventions that are effective are difficult to identify. There is little evidence concerning optimal ways of tailoring interventions for socially disadvantaged groups other than ethnic minorities. A focus on printed information may not provide access to effective methods of information delivery (e.g. tailored information, use of narratives and user generated content). Developing a framework for the effective use of information needs to take account the full range of the factors identified. © 2010 Blackwell Publishing Ltd.

With one million people treated every 36 hours, routinely collected UK National Health Service (NHS) health data has huge potential for medical research. Advances in data acquisition from electronic patient records (EPRs) means such data are increasingly digital and can be anonymised for research purposes. NHS England’s care.data initiative recently sought to increase the amount and availability of such data. However, controversy and uncertainty following the care.data public awareness campaign led to a delay in rollout, indicating that the success of EPR data for medical research may be threatened by a loss of patient and public trust. The sharing of sensitive health care data can only be done through maintaining such trust in a constantly evolving ethicolegal and political landscape. We propose that a dynamic consent model, whereby patients can electronically control consent through time and receive information about the uses of their data, provides a transparent, flexible, and user-friendly means to maintain public trust. This could leverage the huge potential of the EPR for medical research and, ultimately, patient and societal benefit.

Title: Parents of children with ambiguous genitalia: Stories of experiences of reconstructive genital surgeries and finding harmony.\ud \ud Aim: To explore and understand parents' experiences of their child's genital ambiguity and the reconstructive surgeries for ambiguous genitalia that occurred in infancy and middle childhood.\ud \ud Background: The determination of sex and gender for a child born with ambiguous genitalia is an extremely complex medical and social process. Academic debate, professional practice, the law and increased political and ethical debate have all more recently challenged the evidence base for practice. Currently the 'optimal gender policy' and the 'informed consent policy' drive treatment options. Little research has been conducted to understand the significance gender ambiguity has in parents' lives and how the child's genital surgery affects parents.\ud \ud Methods: An exploratory design of narrative inquiry was chosen and data were collected through eighteen in-depth narrative interviews with a purposive non-random sample of fifteen parents of 11 children (aged 0-11 years).\ud \ud Findings: Narrative analysis resulted in three keystone stories which contained in total eight aggregate stories and twenty foundational stories. The three keystone story themes were firstly, parents' stories about their child. Secondly, stories about being a parent of a child with AG and finally stories about healthcare professionals. Interpretation and synthesis of the three keystone stories revealed three core elements fundamental to parents stories; shock protection and anxiety. Parents had to develop new skills in order to deal with the challenges of living with a child with AG. Parents endeavoured to find a sense of harmony from their experiences of shock, anxiety and the need to protect their child. Harmony is a concept that brought consistency and agreement together resulting in parents embracing their experiences holistically and giving their experiences meaning.\ud \ud Conclusion: Parents overcame the tensions inherent in their experience of their child's AG and found a sense of harmony which has not previously been described in the literature.

Dental decay remains a global issue but, in the UK, has become concentrated within the most vulnerable section of society, namely young children and the most deprived. When dental decay and subsequent infection become too severe, teeth may need to be extracted under General Anaesthetic. The aim of this work was to develop a greater understanding of the population of young children referred for a Dental General Anaesthetic (DGA) in the North West of England and to explore possible interventions that may positively impact on this group reducing the high number of children undergoing this procedure. A mixed methods approach was utilised, combining a broad scoping review, quantitative data collection and qualitative interviews. Individually these methods were used to develop a greater understanding of the issue and of the services, processes and patients involved in DGA. The methods were subsequently combined using triangulation to address potential preventative interventions that would be appropriate and acceptable to this group.Differences were observed between hospitals in treatment experiences of children across the North West of England. These included variations in the environment, hospital setting and time from referral to treatment, all of which were shown to impact on the child. The number of children experiencing a repeat DGA was also high and noted as an area that required further intervention. While DGA could be a distressing experience it was considered necessary and resulted in a positive outcome both in terms of oral health and a child’s quality of life. Improving oral health in young children who experience severe decay is a complex and challenging area. A number of interventions were identified that could reduce the need for extraction under DGA. These ranged from targeted interventions, linked to information provision and enablement to support those who are more at risk, to policy change with regard to excessive sugar in food and beverages that could have a broader impact across the population.In addition, DGA service re-designs are discussed including increased provision of prevention that may benefit children referred along this pathway and thereby reduce repeat DGA rates in the future.

Research demonstrates that perspectives of health and illness vary by social and cultural context. This has implications for the ways in which people experience and respond to health and illness and becomes particularly important when people face major social and cultural change through migration. This is explored in this study through the relationship between health and place. The location for the study is a Chinese community centre, in which the centre members are first generation migrants from Hong Kong, China and Vietnam, aged 50 and over, who have spent the larger part of their lives living in the UK. The study uses the concept of therapeutic landscapes as an analytical lens through which to explore understandings of health and illness, issues of identity and belonging, and practices of wellbeing as they are enacted outside of formal healthcare settings.As an ethnographic study, the primary means of data collection has been through participant observation. This included regular attendance at the community centre to participate in activities and events over a period of ten months from August 2013 to May 2014. Twenty one formal interviews were also conducted with members of the community centre, the majority in English, and several in Cantonese. The migration stories of the participants in the study are explored as gendered experiences; that is, that the men and women experienced, and spoke about, migration differently. For the women in particular, their experiences of migration were recalled as a time of profound loneliness and isolation. Understandings of health and illness among the centre members are also explored. A shared understanding of health as a holistic and collective concept was expressed. In particular, they spoke about maintaining a positive attitude in the face of difficulties, about their own health in terms of family and social relationships, and the importance of being together and being active. The choices that they make around the use of Chinese and/or biomedicine are also explored within the context of this understanding. The experiences of migration and the understandings of health and illness are further explored through a consideration of the everyday practices, and associated materialities, that constitute the day-to-day life of the centre. These are explored as ways of re-connecting with the past and maintaining a sense of identity, but also as ways of negotiating both continuity and change at the same time. The role of the community centre in the lives of its members, and the ways in which they interact with one another in this particular place, is approached through the concept of therapeutic landscapes. The day-to-day activities, and the ways in which the centre members participate in these are presented as everyday practices of care; as the enactment of a particular understanding of health and wellbeing that helps to create a sense of identity and belonging at the community centre, which in turn contributes to the health and wellbeing of the centre members.

The Internet is increasingly being used as a source of health advice and information by individuals with long term conditions (LTCs). Online forums allow people to interact with others with similar conditions, providing access to a form of social support that is based around the shared experiences of living with and managing an LTC. However, it is not clear how this support is integrated into people’s lives and their use of health services. To date, research in this area has predominately focused on single conditions, and while it has been suggested that Internet forums may be particularly beneficial for those with ‘contested’ LTCs, this has not been adequately explored. This study aims to address this gap by exploring how individuals with contested and uncontested LTCs utilise Internet forums.This study used qualitative methods, and semi-structured interviews were conducted with 20 participants with ME/CFS and 21 participants with type 1 and 2 diabetes. These conditions were considered to provide an appropriate context in which to explore the use of the Internet and Internet forums for contested and uncontested LTCs. Participants were recruited via a number of online and offline routes, including discussion boards, email lists, newsletters, and face-to-face support groups. Interviews transcripts were analysed using thematic analysis.The results focused on three overlapping themes that were identified during the analysis; namely (1) the process of developing and maintaining trust on forums, (2) accessing support and empowerment online, and (3) forum users’ presentations of self online. The use of online discussion groups was a complex and nuanced process, and was influenced by a number of individual and illness specific factors. Developing relationships with trusted forum members enabled individuals to give and receive support online, and provided a tool of empowerment. While access to peer support and the lived experiences of others were valued by interviewees, forums also allowed participants to access both experiential and scientific knowledge online, providing both lay and expert perspectives around LTCs. In addition, interviewees were seen to carefully manage how they presented themselves on forums, selecting the information that they shared about themselves in order to develop and maintain a particular online persona.By drawing on the advice, information, and support shared online, participants were empowered to position themselves as active participants in their own healthcare and to further engage with healthcare professionals. The findings indicate that forums can play a valuable role in aiding and motivating individuals in the daily management of LTCs, and highlight how this support is used to complement formal health services.

Background: Whilst the experience of chronic physical conditions is well documented and has been recognised as relevant for health policy and practice little is known about notions of recovery and prognosis from the point of view of those with long-term physical health conditions. The extent to which people consider the future outcomes of their conditions is relevant to health policy which seeks to engage people in shared decision making, care plans, and self-management. This gap in knowledge about lay perceptions of recovery and prognosis becomes more pronounced when compared with literature from the mental health field in which recovery is one of the dominant foci, is comparatively well researched and in recent years has fed into policy and management approaches.Aims: The aim of the thesis is to explore perceptions of recovery and prognosis with people with long-term physical health conditions and to compare these with perspectives on recovery and prognosis apparent in the mental health field.Methodology: Using qualitative methods, a two phased approach to data collection and analysis was undertaken. Phase 1 used secondary data analysis with two existing datasets to examine whether notions of recovery and prognosis were implicit in narratives about the experience of illness. Phase 2 built on the findings from phase 1 and utilised longitudinal, primary data collection in the form of narrative interviews undertaken at two time points (baseline and 12 month follow-up). The analysis in both phases involved a cross case thematic analysis to look for commonalities and differences across individuals. Data from phase 2 were also subject to a narrative emplotment of individual stories which were used to capture the longitudinal changes in patient perspectives over time.Results: There were similarities with findings from the mental health field (recovery as a complex, nonlinear journey, the input from friends and family, notions of burden and the impact of condition on sense of self). However, there were nuanced differences in relation to physical health conditions which related to expectations about mortality, the experience of time, the extent to which narratives were future oriented and the experience of stigma. The dual focus on mental and physical health recovery proved useful for understanding those experiences of multiple morbidities. The results were used to develop a model of recovery narratives based on two dimensions (expectations and responsibility) which gave rise to four typologies of narratives. The aim of this model was to further highlight and summarise the themes arising from the data analysis.Discussion: The results of this study highlight the importance of understanding notions of recovery and prognosis in order to better understand the experience of illness and self-management. The thesis challenges the blanket use of health promotion strategies for those with and without chronic health conditions and supports a shift in policy focus from improved choice and autonomy to what Mol (2009) refers to as ‘enhanced care’.

Background: Evidence from the employee perspective frequently suggests that unsupportive managerial relations present a considerable barrier to those with long-term health conditions (LTCs) both on their ability to sustain employment and manage their condition at work. However, little is empirically known about employers’ and managers’ experiences of supporting those with LTCs, or indeed about their perception of their supportive role in the social context of the workplace on which employees suggest they depend for workplace success. This presents a disparity in understanding the contribution of the management role in influencing the (re)entry process to employment for those with LTCs. This is important to explore in light of ongoing objectives by the UK Government to move people with LTCs off incapacity benefits and back into the labour force, as any successful return to work will largely be influenced by employers’ and managers’ readiness to support them.Method: A qualitative approach informed by Grounded Theory principles to guide data collection and analysis was taken for this 3½ year study (September 2009 to March 2013). 40 semi-structured in-depth interviews were conducted with employers and managers from a range of organisations in the North West of England and analysed thematically. Findings were interpreted in relation to a framework of sociological theories of emotion and work. Results: Regardless of industry type, sector and size or condition, several themes emerged which contributed to a sense of burden and tension for participants in supporting those with LTCs. These included discerning legitimacy and tangibility of conditions, having difficult conversations with employees and the influence of the employees’ personality on support. More significantly, all bar one participant typified their role as one of a difficult ‘balancing’ act of additional and often incompatible demands, pressures and feelings arising from managing a complex and emotive situation many considered non-normative to their everyday role. This was typified by feelings of conflict and emotional discomfort, interpreted as ambivalence, stemming from contradictions between and within the normative expectations of their social roles and appropriate feeling rules. For example between the obligations of the professional ‘public’ managerial role in providing value to the organisation, and philanthropic concerns for the welfare of the employee concomitant with their personal ‘private’ role as an individual. Interpreted from a combination of both spoken word and ‘unsaid’ gestures, is the emotion management conducted by managers to cope with negative feelings of ambivalence in a culture which favours rationality over emotionality. Conclusion: Overall, findings indicate that participants in this study concur with the employees’ perspective as to the importance of socially supportive managerial relations. However, it is theorised that managing the emotions of ambivalence serves to undermine manager’s capacity to translate intention to provide support into tangible action, and hence is reflected in the employee’s perception of unsupportive relations. It is suggested that this research could be used to inform the development of a potential intervention to support managers in their pivotal role in the return-to-work process, being beneficial for all stakeholders - the Government, employees and employers alike.

Background: Much social network analysis in health related research has focused on the impact of social networks in the genesis of long term conditions and associated inequalities. However, there has been little research on the role of social networks in the management of such conditions. This is significant at a time when increasing policy emphasis is placed on individual self-care and assumptions are often made about social support, and familial support in particular. The management of chronic illness is complex involving the interplay of personal and contextual factors and comprised of a number of daily activities which include the ongoing negotiation of management into existing roles and the degree of individual engagement with self-management. Little is known about how these aspects interact to shape and influence management, what constitutes a social network for individuals with long term conditions and whether this changes over time.Methods: This study was embedded within the Whole Systems Informing Self-management Engagement (WISE) randomised control trial. It used a longitudinal qualitative design with initial face to face in-depth interviews, telephone follow-ups over a year and final face-to-face interviews where sociograms were also used to elicit network structure.Results: The findings suggest that who is in the social network, and the types of relationships which are present, influence how management practices are framed and the extent to which they are engaged with. Resources available to an individual through the network support, or undermine, engagement and changed over time. Networks included family, friends, GP, nurses and companion animals. The amalgamation of the different types of relationships that constitute the social networks are characterised by three typologies; the family focused network, the friend focused network and the health care professional focused network. These network types reflected where support was sought in times of crisis. In the absence of ‘typical’ sources accounts highlighted the substitutability of network members which was often narrowly sought from key individuals. Changes in illness management, either positive or negative, were framed around critical moments as they challenged existing norms of practice and involved significant network members. Further, co-morbid conditions placed an additional complexity to management and priorities were shaped by the recursive relationship with health services which seemingly influences patients’ orientation in becoming either active assessors of health care or less engaged through being overwhelmed. Conclusions: This thesis challenges the notion of ‘self’-management as an individual construct as many of the practices of illness management involved the support and/or negotiation of roles with others. Critical moments are a key point where normalised management practices are challenged and have particular relevance for future interventions which could foster these critical points to facilitate such changes in routines. Interventions and education need to reflect this wider setting in which chronic illness management occurs for the practices of management to become normalised into everyday routines.

General practice emerged as a distinct medical discipline in the nineteenth century. As independent contractors, General Practitioners (GPs) have however largely been ‘untouched’ by centrally derived policy. As a result, the profession has possessed wide discretion in relation to the way they dealt with their patients. However, due to increasing concerns over the cost and quality of care within the NHS, general practice increasingly became a focal point for the attentions of central policy makers who sought to control aspects of frontline practitioner behaviour. In order to do attempt to align the frontline behaviour of GPs with such policy aims, policy makers turned to their main tool, the contract. In this thesis I am concerned with the most recent contractual changes (and its later variants) introduced in 2004. In particular, the study is concerned with the impact of the large element of Pay-for-performance (P4P) known as the Quality and Outcomes Framework (QOF) contained within the new contract. QOF rewards practices on the basis of meeting a number of targets in relation to clinical, organizational, and patient experience indicators. As a result of the scale and prescriptive nature of the targets, QOF had the potential to change the nature of GP work at the micro-level should GPs choose to follow this voluntary policy. Previous evidence in relation to GP responses to other prescriptive policies such as National Service Frameworks (NSFs) and clinical guidelines suggests that GPs responded as workers, specifically as street-level bureaucrats (SLBs) by selectively by choosing aspects of policies based upon the on the criteria of whether or not they made the practicalities of processing their daily workload easier. However, the evidence suggests that there were also instances of GP principal (those that (part-) own their practices) behaviour that did not conform to expected SLB behaviour but instead resembled behaviour that would be expected of those managers who are ‘results oriented.’ Based upon this evidence and the analytical possibilities the SLB framework provided, the theoretical view of GPs as frontline public sector workers or street-level bureaucrats (SLBs) was employed to understand the continuing perceived impact and responses of GPs to the new contract and in particular QOF. Unlike previous analyses of GPs as SLBs however, this study distinguished between GP principals and salaried GPs employed by the GP principal counterparts. Ultimately, the aim of the thesis was to address the question of whether or not the conceptualization and responses of GPs as SLBs was still relevant and useful post-contractual change. Data was collected (between Feb 2008 and Sept 2009) via semi-structured interviews. In total 62 first round interviews and 24 second round interviews were conducted and analysed thematically. The findings indicate that the financial incentives within the QOF appear to strongly influence the responses of GP principals and reflect their priorities as owners of, rather than workers in their organisations. In addition, it appears that the Evidence Based Practice (EBP) movement means that salaried GPs priorities are also aligned to those of their organisations as they believe most of the QOF to be evidence-based. As a result, the application of Lipsky’s SLB framework to explaining GP behaviour in relation to QOF is less useful than previous applications.