Experiences of young people growing up in a family with Huntington's disease: A meta-ethnography of qualitative research.

Huntington's disease is a genetic neurodegenerative condition with wide physical and psychological impacts. Children of a parent with the condition have a 50% chance of carrying the gene expansion and developing the condition themselves. This systematic review and meta-ethnography presents a synthesis of the qualitative research on the experiences of young people growing up in a family with Huntington's disease. The MEDLINE, PsycINFO, and CINAHL databases were systematically searched, and 13 papers met the inclusion criteria. Through the process of meta-ethnography, four themes were identified highlighting aspects of childhood that were stolen and fought for: thief of relationships, thief of self, thief of transparency, and search for reclamation. Within the themes, the complex challenges young people faced when growing up in a HD family were explored such as the impact of adverse childhood experiences and the possible effects of HD on attachment and social relationships. Clinical implications are considered, and recommendations are made for future research.

Living with Parkinson's in England during and beyond COVID-19 restrictions: a longitudinal qualitative study.

OBJECTIVES: Government-enforced lockdown restrictions associated with preventing the spread of the COVID-19 virus had a series of unintended, negative effects. One group of individuals whose physical and mental health was significantly and disproportionately impacted were those with Parkinson's. However, research has been mainly cross-sectional, with no previous study qualitatively following up participants through both lockdowns and the easing of restrictions. Consequently, this study aimed to provide a detailed understanding of the experience of lockdowns and the easing of restrictions on the physical and mental health of people with Parkinson's. METHOD: Data from semi-structured interviews collected at four time points across an 18-month period (May 2020 - September 2021) from the same participants (six men and four women) were analysed using interpretative phenomenological analysis. RESULTS: Three themes were derived: (1) Wrestling with a Parkinson's identity, agency and control during the pandemic; (2) The encroachment and acceleration of a Parkinson's future; and (3) Recalibrating priorities from COVID-19 to Parkinson's. CONCLUSION: As currently the only published study to provide an in-depth longitudinal analysis with this population, we used a more dynamic theoretical account, Strauss and Corbin's theory of illness trajectories, to understand the findings and suggest ways of supporting individuals with Parkinson's in this stage of the pandemic. The scale and breadth of the support needed is a significant challenge for current statutory systems.

Stigma, coping strategies, distress and wellbeing in individuals with cervical dystonia: a cross-sectional study.

Cervical dystonia (CD) is a movement disorder which causes sustained muscle contractions in the neck leading to abnormal postures and repetitive movements. As it is a highly visible condition, people with CD can experience stigma, which may lead to unhelpful coping strategies and increased psychological distress. This study investigated whether adaptive and maladaptive coping strategies mediate the relationship between stigma and psychological outcomes in people with CD. A total of 114 adults with CD completed measures of stigma, coping, health-related quality of life (HRQOL), psychological distress (depression, anxiety, stress), and psychological wellbeing at one time point. Participants' levels of distress were high, compared to the general population. Correlational analyses showed increased stigma and maladaptive coping (e.g. substance use, behavioural disengagement) were both significantly related to increased distress, lower wellbeing and lower HRQOL, whereas higher adaptive coping (e.g. acceptance, humour) was only related to higher wellbeing. In a parallel mediation model, maladaptive coping strategies mediated the relationship between stigma and distress, HRQOL and wellbeing, but adaptive coping strategies did not. These findings suggest that maladaptive coping may play an important role in explaining the relationship between stigma and some aspects of distress and wellbeing in CD. Interventions which focus on reducing different aspects of maladaptive coping may be helpful to improve wellbeing as well as reducing stigma.

Neurologists' lived experiences of communicating the diagnosis of a motor neurodegenerative condition: an interpretative phenomenological analysis.

BACKGROUND: Receiving the diagnosis of a motor neurodegenerative condition (MNDC) can be a life-changing experience. Although several studies of individuals' experiences have indicated dissatisfaction with aspects of how an MNDC diagnosis was communicated, few studies have addressed doctors' experiences of breaking bad news for these conditions, especially from a qualitative perspective. This study explored UK neurologists' lived experience of delivering an MNDC diagnosis. METHODS: Interpretative phenomenological analysis was used as the overarching method. Eight consultant neurologists working with patients with MNDCs took part in individual, semi-structured interviews. RESULTS: Two themes were constructed from the data: 'Meeting patients' emotional and information needs at diagnosis: a balancing act between disease, patient and organization-related factors', and 'Empathy makes the job harder: the emotional impact and uncovered vulnerabilities associated with breaking bad news'. Breaking the news of an MNDC diagnosis was challenging for participants, both in terms of achieving a patient-centred approach and in terms of dealing with their own emotions during the process. CONCLUSIONS: Based on the study's findings an attempt to explain sub-optimal diagnostic experiences documented in patient studies was made and how organizational changes can support neurologists with this demanding clinical task was discussed.

Parkinson's and the couple relationship: a qualitative meta-synthesis.

OBJECTIVE: The aim was to synthesise the current qualitative literature on the impact of Parkinson's on the couple relationship, including individual and dyad studies. METHODS: Noblit and Hare's meta-ethnography approach was applied; 19 studies were included in the review following a systematic search of four electronic databases. The studies included experiences of 137 People with Parkinson's and 191 partners. FINDINGS: Analysis produced three themes: (1) Disruption of roles and responsibilities; (2) Challenges to communication and closeness; and (3) Grief, burden, and isolation. The themes are discussed with supporting extracts from the 19 included studies. CONCLUSION: The findings highlight the challenges that couples experience and the individual and relational resources that support coping. Support should be individually tailored to each couple as the impact on the couple may change in response to individual and contextual factors. This review adds further evidence to the case for relationally focused multidisciplinary team input at all stages of Parkinson's disease.

The experience of a sample of individuals in the United Kingdom living in the pre-manifest stage of Huntington's disease: An interpretative phenomenological analysis.

This paper explores the experience of ten individuals living in the UK who were in the pre-manifest stage of Huntington's disease (HD), a genetic neurodegenerative condition. Data were gathered using semi-structured interviews and analyzed using interpretative phenomenological analysis. Three themes were reported from the data: 'feeling limited by time', 'the perception of stalling time', and 'making the most of time', all highlighting the way in which time holds significant meaning when living in the pre-manifest stage of HD. This study has highlighted the difficulties experienced by individuals when adjusting to the pre-manifest stage of HD. Feeling able to manage their anxieties and dealing with ongoing uncertainty related to future deterioration was key to supporting their wellbeing, facilitated by factors such as positivity and hope. Individuals may benefit from counseling which supports them to develop proactive coping strategies to manage their anxieties and acceptance of an uncertain future.

Neurologists' current practice and perspectives on communicating the diagnosis of a motor neurodegenerative condition: a UK survey.

BACKGROUND: The communication of a life-changing diagnosis can be a difficult task for doctors with potential long-term effects on patient outcomes. Although several studies have addressed the experiences of individuals with motor neurodegenerative diseases in receiving this diagnosis, a significant research gap exists regarding professionals' perspectives, especially in the UK. This study aimed to assess UK neurologists' current practice and perspectives on delivering the diagnosis of a motor neurodegenerative disease, explore different aspects of the process and detail the potential challenges professionals might face. METHODS: We conducted an anonymised online survey with 44 questions, grouped into four sections; basic demographic information, current practice, the experience of breaking bad news and education and training needs. RESULTS: Forty-nine professionals completed the survey. Overall, participants seemed to meet the setting-related standards of good practice; however, they also acknowledged the difficulty of this aspect of their clinical work, with about half of participants (46.5%) reporting moderate levels of stress while breaking bad news. Patients' relatives were not always included in diagnostic consultations and participants were more reluctant to promote a sense of optimism to patients with poorer prognosis. Although professionals reported spending a mean of around 30-40 min for the communication of these diagnoses, a significant proportion of participants (21-39%) reported significantly shorter consultation times, highlighting organisational issues related to lack of capacity. Finally, the majority of participants (75.5%) reported not following any specific guidelines or protocols but indicated their interest in receiving further training in breaking bad news (78.5%). CONCLUSIONS: This was the first UK survey to address neurologists' practice and experiences in communicating these diagnoses. Although meeting basic standards of good practice was reported by most professionals, we identified several areas of improvement. These included spending enough time to deliver the diagnosis appropriately, including patients' relatives as a standard, promoting a sense of hope and responding to professionals' training needs regarding breaking bad news.

Psychological inflexibility and somatization in nonepileptic attack disorder.

BACKGROUND: There is no clear understanding of what causes and maintains nonepileptic attack (NEA) disorder (NEAD), or which psychological therapies may be helpful. The relationships between variables of psychological inflexibility: experiential avoidance (EA), cognitive fusion (CF), mindfulness, and key outcome variables in NEAD: somatization, impact upon life, and NEA frequency were investigated. METHOD: Two hundred eighty-five individuals with NEAD completed validated measures online. Linear regression was used to explore which variables predicted somatization and impact upon life. Ordinal regression was used to explore variables of interest in regard to NEA frequency. RESULTS: Mindfulness, EA, CF, somatization, and impact upon life were all significantly correlated. Mindfulness uniquely predicted somatization when considered in a model with EA and CF. Higher levels of somatization increased the odds of experiencing more NEAs. Individuals who perceived NEAD as having a more significant impact upon their lives had more NEAs, more somatic complaints, and more EA. CONCLUSIONS: Higher levels of CF and EA appear to be related to lower levels of mindfulness. Lower levels of mindfulness predicted greater levels of somatization, and somatization predicted NEA frequency. Interventions that tackle avoidance and increase mindfulness, such as, acceptance and commitment therapy, may be beneficial for individuals with NEAD. Future directions for research are suggested as the results indicate more research is needed.

Self-compassion and depression, anxiety, and resilience in adults with epilepsy.

BACKGROUND: Research suggests that people with epilepsy (PWE) are more likely to experience depression and anxiety than the general population. Given the adversity associated with the condition, resilience may also be important. However, to date, resilience has been largely overlooked in the epilepsy literature. Self-compassion has been widely associated with improved psychological wellbeing and, to a lesser extent, resilience. However, the relationship between self-compassion and depression, anxiety, and resilience in PWE has not been examined. OBJECTIVES: Using a quantitative cross-sectional survey design, the aim of the present study was to examine the extent to which self-compassion predicted depression, anxiety, and resilience when controlling for demographic and illness-related variables. METHODS: Adults with epilepsy were invited to take part in a survey online or in epilepsy or neurology clinics. Two-hundred and seventy participants completed the survey, and data were analyzed using hierarchical multiple regression models. RESULTS: In this sample of PWE, self-compassion significantly predicted lower depression and anxiety and higher resilience when other significant sociodemographic and illness-related variables had been taken into account. CONCLUSIONS: The findings of the present study indicate that self-compassion could be an important factor in determining psychological outcomes for adults with epilepsy, and its role is worthy of further exploration to help improve psychological outcomes for PWE.

Avoidance in nonepileptic attack disorder: A systematic review and meta-analyses.

BACKGROUND: Avoidance is the active process of trying to escape from or not experience situations, places, thoughts, or feelings. This can be done through behavioral or cognitive strategies, or more broadly, a combination of both, utilized in an attempt to disengage from private experiences referred to as experiential avoidance (EA). Avoidance is considered important in the development and maintenance of nonepileptic attack disorder (NEAD). This review aimed to understand avoidance in NEAD and evaluate its role as a contributory factor. METHODS: Fourteen articles were identified by searching Cumulative Index to Nursing and Allied Health (CINAHL), MEDLINE Complete, PsycINFO, and EMBASE and were combined in a narrative synthesis. Six of these articles were included in a meta-analysis comparing levels of EA for individuals with NEAD and healthy controls (HC), and four were included in a meta-analysis comparing EA in NEAD to epilepsy comparisons (EC). CONCLUSIONS: Experiential avoidance appears to be a strategy that is used by a high proportion of the population with NEAD. The group with NEAD utilized significantly more avoidance compared with both the HC and EC. However, further research is needed to understand the extent and types of avoidance that are relevant.

The views of adults with neurodegenerative diseases on end-of-life care: a metasynthesis.

OBJECTIVE: Improving end-of-life care for people with neurodegenerative diseases is seen as a clinical priority. In order to do this, it is important to take into account the views expressed by people with these conditions on their experiences of this care. The purpose of this review was to provide a thematic synthesis of the views of adults with neurodegenerative diseases on end-of-life care. METHODS: After a systematic search, 13 articles were included and thematic synthesis was used to collate and interpret findings. RESULTS: Four analytical themes were identified; (1) Importance of autonomy and control; (2) Informed decision-making and the role of healthcare professionals; (3) Contextual factors in decision-making; (4) The pitfalls of care. CONCLUSION: Participants' views were framed by the context of their lives and experience of their illness and these shaped their engagement with end-of-life care. Given the varying disease trajectories, care needs to be individualised and needs-based, implementing palliative care in a timely way to prevent crises and loss of autonomy.

Correlates of stigma in adults with epilepsy: A systematic review of quantitative studies.

OBJECTIVES: The aim of this review was to identify quantitative correlates, predictors, and outcomes of stigma in adults with epilepsy living in Western countries. METHODS: To identify relevant literature, four academic databases (PsycINFO, CINAHL, PubMed, and Scopus) were systematically searched using key terms related to stigma and epilepsy. RESULTS: Thirty-three research papers reporting findings from 25 quantitative studies of correlates of stigma in epilepsy were identified. The findings suggest that stigma can be predicted by demographic, illness-related, and psychosocial factors, although associations were found to be highly culturally specific. Outcomes of stigma in people with epilepsy were replicated more consistently across cultures, and its impact was significant. Detrimental effects included both worse physical health, including less effective management of the condition, and reduced psychological well-being, including difficulties such as depression and anxiety. IMPLICATIONS: Educational initiatives and therapeutic interventions that aim to address stigma in people with epilepsy are recommended; however, these need to be culturally informed to ensure that they are valid and effective.

The views of adults with Huntington's disease on assisted dying: A qualitative exploration.

BACKGROUND: Assisted dying is frequently debated publicly and research often includes the views of health professionals on this issue. However, the views of people with life-limiting conditions, for whom this issue is likely to have a different resonance, are less well represented. AIM: The purpose of this study was to explore the views of people who live with the inevitability of developing Huntington's disease, a genetically transmitted disease which significantly limits life, on assisted dying. DESIGN: Using thematic analysis methodology, individual semi-structured interviews were conducted. SETTING/PARTICIPANTS: Seven participants (five women and two men) who were gene positive for Huntington's disease took part in the study. RESULTS: Four themes were extracted: (1) autonomy and kindness in assisted dying: the importance of moral principles; (2) Huntington's disease threatens life and emphasises issues relating to death; (3) dilemmas in decision-making on assisted dying: "There are no winners" and (4) the absence of explicit discussion on dying and Huntington's disease: "Elephants in the room". CONCLUSIONS: Our findings suggest that talking to patients about assisted death may not cause harm and may even be invited by many patients with Huntington's disease. The perspectives of those who live with Huntington's disease, especially given its extended effects within families, add significant clinical and theoretical insights.

The experiences of parenting a child with an acquired brain injury: A meta-synthesis of the qualitative literature.

OBJECTIVE: To systematically review and then synthesize the qualitative literature on the experience of parenting a child with an acquired brain injury (ABI). DESIGN: Systematic literature review and meta-synthesis Methods. A systematic search of the literature was conducted in four databases. Papers which met the inclusion criterion were assessed for quality using the Critical Skills Appraisal Programme (CASP) tool and then synthesized according to Noblit and Hare's (1988) guidelines for meta-ethnography. RESULTS: Of the 4855 papers retrieved, 17 met the inclusion criteria. Synthesis resulted in three themes: (1) Disconnection: Cut off from internal emotions and isolated from others; (2) Seeking understanding and support to manage in an insecure world; and (3) New parent to a different child. CONCLUSIONS: Having a child with an ABI leads to many challenges for parents. These include feeling insecure, isolated from others and struggling to adapt to the different roles required to parent their different child. Clinical implications highlight the need for specialist support that is ongoing after discharge, including specialist knowledge and understanding of ABI and opportunities for peer support.

The experience of caring for patients with dementia within a general hospital setting: a meta-synthesis of the qualitative literature.

OBJECTIVES: The optimal care of people with dementia in general hospitals has become a policy and practice imperative over recent years. However, despite this emphasis, the everyday experience of staff caring for this patient group is poorly understood. This review aimed to synthesise the findings from recent qualitative studies in this topic published prior to January 2014 to develop knowledge and provide a framework to help inform future training needs. METHOD: A systematic search of the literature was conducted across five academic databases and inclusion/exclusion criteria applied to the retrieved papers. A meta-ethnographic approach was utilised to synthesise the resulting 14 qualitative papers. RESULTS: Five key themes were constructed from the findings: overcoming uncertainty in care; constraints of the environmental and wider organisational context; inequality of care; recognising the benefits of person-centred care; and identifying the need for training. These themes explore the opportunities and challenges associated with caring for this group of patients, as well as suggestions to improve staff experiences and patient care. CONCLUSION: The synthesis highlighted a lack of knowledge and understanding of dementia within general hospital staff, particularly with regard to communication with patients and managing behaviours that are considered challenging. This limited understanding, coupled with organisational constraints on a busy hospital ward, contributed to low staff confidence, negative attitudes towards patients with dementia and an inability to provide person-centred care. The benefits of dementia training for both ward staff and hospital management and peer discussion/support for ward staff are discussed.

Same-sex partner bereavement in older women: an interpretative phenomenological analysis.

OBJECTIVES: Due to the lack of existing literature, the current research explored experiences of same-sex partner bereavement in women over the age of 60. METHOD: Semi-structured interviews were conducted with eight women. Transcripts were analysed using interpretative phenomenological analysis. RESULTS: Three themes were identified which elaborated the experiences of older women who had lost a same-sex partner: (1) being left alone encapsulated feelings of isolation and exclusion; (2) navigating visibility centred on how homophobia led to a lack of recognition of the women's grief; and (3) finding new places to be authentic related women's need for new relationships in which they could be themselves. The findings indicate that existing models of partner bereavement may provide useful frameworks when seeking to understand the experiences of older women who have lost their same-sex partners. CONCLUSION: The findings indicate that in addition to the experiences of partner bereavement noted in research with heterosexual widows, older women who lose same-sex partners may face particular challenges, which can impact upon psychological well-being and adjustment to loss. These challenges appear to result from past and current homophobic and heterosexist attitudes within the UK culture. A range of interventions at individual, group, health service, and societal levels may be beneficial in improving the psychological well-being of older women who lose a same-sex partner.

Coping with Tourette's syndrome: a meta-ethnography of individual and family perspectives.

OBJECTIVE: This systematic review and meta-ethnography aimed to examine how children, adults and families cope with Tourette's syndrome (TS). METHODS: A systematic search of four databases was completed in October 2022. Sixteen papers met the inclusion criteria and were synthesised using Noblit and Hare's (1988) meta-ethnographic approach. RESULTS: Three themes were constructed: redefining the self and social identity, controlling the visible presentation of Tourette's syndrome, and challenging the narrative. CONCLUSION: Findings indicate that coping involves the need to integrate TS with identity, to exert control over tics and to challenge the misrepresentations of TS in wider society. A supportive environment provided by parents and friends enables individuals to feel proud that they can control their tics, and this allows for the positive integration of TS into identity. Raising awareness at a societal level through educational campaigns is important when aiming to improve coping with a stigmatised condition. Further research is recommended, for example, to understand how common co-occurring conditions, such as attention deficit hyperactivity disorder, impact coping.

Stigma, self-compassion, and psychological distress among people with Parkinson's.

PURPOSE: People with Parkinson's disease (hereafter Parkinson's) can experience stigma through the attitudes and actions of others (enacted stigma) and through anticipation of enacted stigma and internalisation of negative stereotypes (felt stigma). Self-compassion may protect against the impact of stigma. This study aimed to investigate the relationships between self-compassion, stigma, and psychological distress among people with Parkinson's. METHODS: A total of 130 people with Parkinson's completed questionnaires measuring self-compassion, enacted and felt stigma, and depression, anxiety, and stress. Correlation, mediation, and moderation models were used to investigate relationships between variables. RESULTS: All variables correlated significantly in the expected directions. Felt stigma mediated the relationship between self-compassion and the three outcome variables - depression, anxiety, and stress. Self-compassion did not moderate the relationship between enacted stigma and distress and suggested enacted stigma was associated with distress, regardless of levels of self-compassion. CONCLUSIONS: Self-compassion and both enacted and felt stigma are important predictors of distress for people with Parkinson's. Part of the relationship between lower self-compassion and psychological distress appears to occur via the internalisation of stigma. These findings may be relevant to the development of individualised and societal interventions with the aim of improving the psychological wellbeing of people with Parkinson's.Implications for rehabilitationSelf-compassion was associated with lower levels of psychological distress (i.e., depression, anxiety, and stress) and self-stigma partially mediated this relationship.Self-compassion did not moderate the relationship between enacted stigma and psychological distress, suggesting enacted stigma increases distress, regardless of self-compassion.The development and assessment of the effectiveness of compassion-focused interventions tailored for people with Parkinson's may be important as well as systemic stigma focused interventions.

The joint impact of symptom deterioration and social factors on wellbeing for people with Parkinson's during the covid-19 pandemic in the UK.

The covid-19 pandemic and associated restrictions have had significant consequences for those living with chronic conditions such as Parkinson's. The restrictions in access to healthcare as well as reductions in social care, family support and community activities have led to decreases in physical and mental wellbeing. However, not everyone has been equally affected and the predictors of distress are currently being investigated worldwide. Here we use data from a UK survey conducted by the charity Parkinson's UK during Summer 2021 to look at physical and social predictors of wellbeing of people with Parkinson's. Specifically, we aimed to look at the combined effects of worsening physical symptoms, social isolation and loneliness on psychological wellbeing when controlling for age, gender and disease duration. The data from 612 participants were analysed using multiple regression analyses and showed that worsened physical symptoms, loneliness and social isolation each independently predicted wellbeing thus showing the impact of both physical symptoms and social factors. Improved access to healthcare and physical activity is needed to help improve physical health. However, addressing the social needs of people with Parkinson's is also important, and not only during a pandemic. Additional interventions may be needed to reduce social isolation and loneliness as there may be added barriers for people with Parkinson's which need to be considered.

Guided self-help for anxiety among Huntington's disease gene expansion carriers (GUIDE-HD) compared to treatment as usual: a randomised controlled feasibility trial.

BACKGROUND: Huntington's disease (HD) is an adult-onset genetic neurodegenerative condition associated with cognitive decline, motor impairments, and emotional difficulties. Anxiety affects up to 71% of HD gene expansion carriers (i.e., those with the version of the gene that causes HD) and can negatively impact quality of life, worsen other HD symptoms, and increase suicide risk. Therefore, helping people with their anxiety should be a clinical priority. A significant evidence base now exists for low-cost talking therapies for anxiety, such as guided self-help, and with people with other neurodegenerative conditions (e.g., Parkinson's disease). However, this type of intervention has not been specifically assessed with HD gene expansion carriers. METHODS: This protocol describes an exploratory randomised controlled feasibility study of a psychological intervention for anxiety for HD gene expansion carriers. The 10 session guided self-help intervention ('GUIDE-HD') is based on a blend of second and third wave cognitive behavioural models of anxiety (cognitive behaviour therapy [CBT] and acceptance and commitment therapy [ACT]) and is adapted to meet the specific needs of an HD population. This study will compare guided self-help with treatment as usual (TAU), with 15 HD gene expansion carriers randomly allocated to each group. Participants will be recruited across the UK. Quantitative data will be collected pre-intervention, immediately post-intervention, 3-month post-intervention and 6-month post-intervention. Qualitative data will be collected at one month post-intervention from participants, including HD carers. The data will be analysed to assess whether the current intervention and study design are feasible to progress to a larger randomised controlled trial. Feasibility has been defined in terms of recruitment rate, retention rate to both trial arms, intervention adherence, and acceptability of the intervention and measurement tools. DISCUSSION: Given the lack of evidenced interventions to date to support the wellbeing of people with the expanded Huntington's gene, this study will assess the feasibility of progressing this particular intervention to a full trial. To try and increase the acceptability of the intervention, a number of stakeholders, including those affected by HD and in caring roles, have been fundamental to the creation of the intervention (e.g., therapy manual, planned therapy process) to date. TRIAL REGISTRATION: Trial ID: ISRCTN47330596 . Date registered: 28/09/2022. Protocol version and date: Version 2, 09/06/22. Trial sponsor organisation and contact: Leicestershire Partnership NHS Trust (Dave Clarke). Role of sponsor: Overall responsibility for the conduct and governance of the trial. Role of funder: Review of initial research proposal.