Parental concern for clinically vulnerable child during first 18 months of the COVID pandemic.

BACKGROUND: The uncertainties surrounding the COVID-19 pandemic have been associated with increased parental concern. The aim of this study is to explore if this increased level of concern is associated with certain individual/household characteristics or if parents adapted to the ever-changing realities of the COVID-19 pandemic over time. METHODS: This prospective study explored COVID-19 concern trajectories and associated family characteristics of 765 UK parents caring for an immunosuppressed child during the first 18 months of the pandemic using growth mixture modelling. Qualitative analysis was performed to examine in more detail the source of concern. RESULTS: Four different trajectories of parental COVID-19 concern were identified. Ongoing very high concern was associated with caring for children with nephrotic or respiratory disease; having a child on an organ transplant waiting list; residency in the North of England; or parental vocational inactivity. Explicit concerns voiced by the parents generally followed national trends, but vulnerable status specific concerns were also reported. CONCLUSION: Diagnosis and prescribed medication of the immunosuppressed child, geographical location, household composition, and employment status of parent were associated with the different concern trajectories. This information can be helpful in targeting psychological family care where it is most needed. IMPACT: Many British parents caring for a clinically vulnerable child during the first 18 months of the COVID-19 pandemic showed high levels of concern with little sign of psychological adaptation. Consistent with findings from non-vulnerable populations, parents mentioned the impact of shielding and repeated isolation on their child's education, social life, and mental health. Unique to the clinically vulnerable population, parents were worried about child's health status, impact of delayed healthcare, and were confused by the contradictory information received from government, doctors, and media. Psychological family care can be targeted to those parents at greater risk for high levels of concern.

First validation study of the living with long term conditions scale (LwLTCs) among English-speaking population living with Parkinson's disease.

INTRODUCTION: Parkinson's disease is the second most prevalent neurodegenerative disease, affecting 10 million people worldwide. Health and social care professionals need to have personalised tools to evaluate the process of living with Parkinson's disease and consequently, plan individualised and targeted interventions. Recently, the English version of the Living with Long term conditions (LwLTCs) scale has been developed filling an important gap related to person-centred tools to evaluate the process of living with long term conditions among English-speaking population. However, no validation studies for testing its psychometric properties have been conducted. AIM: To analyse the psychometric properties of the LwLTCs scale in a wide English-speaking population living with Parkinson's disease. METHODS: Validation study, with an observational and cross-sectional design. The sample was composed of individuals living with Parkinson's disease from non-NHS services in the community. Psychometric properties including feasibility and acceptability, internal consistency, reproducibility, and construct, internal and known-groups validity were tested. RESULTS: A total sample of 241 people living with Parkinson's disease were included. 6 individuals did not complete 1 or 2 items on the scale. Ordinal alpha was 0.89 for the total scale. The intraclass correlation coefficient for the total scale was 0.88. The LwLTCs scale is strongly correlated with scales measuring satisfaction with life (rs=0.67), quality of life (rs=0.54), and moderately correlated with social support (rs=0.45). Statistically significant difference just for therapy and co-morbidity, yet no for gender, employment situation, or lifestyle changes. CONCLUSIONS: The LwLTCs scale is a valid scale to evaluate how the person is living with Parkinson's disease. Future validation studies to prove the repeatability of the total scale and particularly, domains 3-Self-management, and 4-Integration and internal consistency will be needed. Developing further studies on the English version of the LwLTC in people with other long term conditions is also proposed.

Health and Social Care Diversity Among Individuals with Longstanding Physical and Psychological Health Problems: Pooled Repeated Cross Sectional Analyses.

This paper examines differences in health-and-social care utilisation for individuals with physical and/or mental health problems. Logistic regression models are used to determine disparity in the percentage of General Household/Lifestyle Survey participants with physical compared to mental health problems receiving disability benefits or health care services between 2000 and 2011. Our findings of a relative underutilisation of secondary health care combined with a relative overutilization of out-of-work benefits by individuals with mental health problems is novel to the field of rehabilitative health care. These results provide evidence for the previously suspected disparity in health care utilisation of individuals with mental health problems and indicate problems in labour force integration. The findings support the political call for a 'parity of esteem', which, in Britain, was enshrined in the Health and Social Care Act of 2012.

Extracurricular activity participation moderates impact of family and school factors on adolescents' disruptive behavioural problems.

BACKGROUND: The prevalence of problem behaviours among British adolescents has increased in the past decades. Following Erikson's psychosocial developmental theory and Bronfenbrenner's developmental ecological model, it was hypothesized that youth problem behaviour is shaped in part by social environment. The aim of this project was to explore potential protective factors within the social environment of British youth's for the presentation of disruptive behavioural problems. METHOD: This study used secondary data from the Longitudinal Study of Young People in England, a cohort study of secondary school students. These data were analysed with generalized estimation equations to take the correlation between the longitudinal observations into account. Three models were built. The first model determined the effect of family, school, and extracurricular setting on presentation of disruptive behavioural problems. The second model expanded the first model by assuming extracurricular activities as protective factors that moderated the interaction between family and school factors with disruptive behavioural problems. The third model described the effect of prior disruptive behaviour on current disruptive behaviour. RESULTS: Associations were found between school factors, family factors, involvement in extracurricular activities and presence of disruptive behavioural problems. Results from the second generalized estimating equation (GEE) logistic regression models indicated that extracurricular activities buffered the impact of school and family factors on the presence of disruptive behavioural problems. For instance, participation in sports activities decreased the effect of bullying on psychological distress. Results from the third model indicated that prior acts of disruptive behaviour reinforced current disruptive behaviour. CONCLUSION: This study supports Erikson's psychosocial developmental theory and Bronfenbrenner's developmental ecological model; social environment did influence the presence of disruptive behavioural problems for British adolescents. The potential of extracurricular activities to intervention strategies addressing disruptive behavioural problems of adolescents is discussed.

Silymarin (Milk Thistle) Treatment of Adults With Gambling Disorder: A Double-Blind, Placebo-Controlled Trial.

OBJECTIVE: Data on the pharmacological treatment of gambling disorder are limited. Silymarin (derived from milk thistle) has antioxidant properties. The goal of the current study was to determine the efficacy and tolerability of silymarin in adults with gambling disorder. METHODS: Forty-three individuals (18 [41.9%] women; mean age=49.61 [±13.1] years) with gambling disorder entered an 8-week, double-blind, placebo-controlled study. Dosing of silymarin ranged from 150 to 300 mg twice a day. The primary outcome measure was the Yale Brown Obsessive Compulsive Scale Modified for Pathological Gambling (PG-YBOCS). Secondary outcome measures comprised the Gambling Symptom Assessment Scale and measures of depression and anxiety. Outcomes were examined using mixed-effect models. RESULTS: Silymarin did not statistically differentiate from the placebo on any of the outcome measures of interest, in terms of treatment group×time interactions. There was a robust response in the placebo group (57% reduction on the PG-YBOCS), and on average there was a 56% reduction in YBOCS score for the milk thistle. CONCLUSIONS: The findings of this study do not support the use of silymarin/milk thistle in the treatment of gambling disorder but highlight the large placebo response seen in gambling disorder. Treatment interventions for gambling disorder need to better understand and address the placebo response. TRIAL REGISTRATION: ClinicalTrials.gov identifier: NCT02337634.

Psychometric properties of the living with long term conditions scale in an English-speaking population living with long term conditions in the UK.

OBJECTIVE: To present the psychometric properties of the living with long-term condition (LwLTCs) scale in an English-speaking population of people with different LTCs. DESIGN: An observational and cross-sectional study, with retest was conducted. Psychometric properties including feasibility, internal consistency, confirmatory factor analysis, reproducibility and content validity were tested. SETTING: The study took place across the UK via primary care surgeries and voluntary organisations, between December 2021 and June 2022. PARTICIPANTS: The study included 577 patients living with different LTCs, as chronic obstructive pulmonary disease, arthritis, chronic heart failure, Parkinson's disease, chronic kidney disease and type 2 diabetes mellitus. Inclusion criteria included: (a) having been diagnosed with one or more of the conditions; (b) being able to read, understand and answer written questionnaires; (c) being fluent in English and (d) being able to provide written informed consent. Patients were involved in the design and pilot study of the scale. RESULTS: A total sample of 577 people with an age range of 37-97 years (98±9.65) were recruited. Internal consistency of the total 26-item LwLTCs scale score was excellent (ordinal alpha=0.90) but confirmatory factor analysis showed better fit indices (Normed Fit Index=0.96; standardised root mean square residual=0.051; Goodness of Fit Index=0.98) for a 20-item LwLTCs scale. CONCLUSIONS: A shorter version of the LwLTCs scale, with just 20 items and with excellent psychometric properties, is recommended. Having a short scale is key when considering the implementation of the scale in clinical practice to develop person-centred pathways and more comprehensive care plans.

Psychological distress experienced by parents caring for an immunosuppressed child during the COVID-19 pandemic.

The COVID-19 pandemic has proved unique in both its unpredictability and the extent to which it has continued to impact on daily life since March 2020. Among the immunosuppressed population the challenges of the COVID-19 pandemic are cumulative to the ever-present challenges of living with a long-term condition. This prospective longitudinal study explored patterns of concern experienced by 467 British parents caring for an immunosuppressed child during the first 2 years of the COVID-19 pandemic and related this to parental mental wellbeing. Most parents slowly adapted or were resilient to the ever-changing stressors of the COVID-19 pandemic. However, 12% experienced high levels of concern throughout the first 2 years of the pandemic. This group was also more likely to report emotional mental health problems towards the end of this period. The experience of emotional mental health problems among parents caring for an immunosuppressed child was related to low household income, single parenting, difficult access to greenspace, and higher level of exposure to COVID positive cases and COVID restrictions (North of England). Parents reported that optimism, reduction of isolation, and support promoted coping and management of the challenges of the COVID-19 pandemic. More reliable COVID information and periodic medical-condition-specific guidance would have been appreciated. These findings can increase clinical awareness of high-risk parental groups and make an important contribution to the planning of appropriate targeted psychological family interventions.

The Palestinian primary ciliary dyskinesia population: first results of the diagnostic and genetic spectrum.

Background: Diagnostic testing for primary ciliary dyskinesia (PCD) started in 2013 in Palestine. We aimed to describe the diagnostic, genetic and clinical spectrum of the Palestinian PCD population. Methods: Individuals with symptoms suggestive of PCD were opportunistically considered for diagnostic testing: nasal nitric oxide (nNO) measurement, transmission electron microscopy (TEM) and/or PCD genetic panel or whole-exome testing. Clinical characteristics of those with a positive diagnosis were collected close to testing including forced expiratory volume in 1 s (FEV1) Global Lung Index z-scores and body mass index z-scores. Results: 68 individuals had a definite positive PCD diagnosis, 31 confirmed by genetic and TEM results, 23 by TEM results alone, and 14 by genetic variants alone. 45 individuals from 40 families had 17 clinically actionable variants and four had variants of unknown significance in 14 PCD genes. CCDC39, DNAH11 and DNAAF11 were the most commonly mutated genes. 100% of variants were homozygous. Patients had a median age of 10.0 years at diagnosis, were highly consanguineous (93%) and 100% were of Arabic descent. Clinical features included persistent wet cough (99%), neonatal respiratory distress (84%) and situs inversus (43%). Lung function at diagnosis was already impaired (FEV1 z-score median -1.90 (-5.0-1.32)) and growth was mostly within the normal range (z-score mean -0.36 (-3.03-2.57). 19% individuals had finger clubbing. Conclusions: Despite limited local resources in Palestine, detailed geno- and phenotyping forms the basis of one of the largest national PCD populations globally. There was notable familial homozygosity within the context of significant population heterogeneity.

The Impact on Parents of Diagnosing PCD in Young Children.

Primary ciliary dyskinesia (PCD) is an incurable, rare, inherited, chronic condition. Treatment includes the regular clearing of airway mucus, aggressive treatment of infections and management of hearing loss. Caregiver burden has not been explored, hence we interviewed 18 mothers and 6 fathers of children under 6 years to understand the impact of diagnostic testing and implications of a positive diagnosis. Interviews were transcribed and thematically analysed and five key themes were identified. These included the parents' experiences following child's diagnosis, impact of child's treatment regimen on parent, impact of child's health status on parent, parent's coping strategies, and parental concerns for the future. Parents described their diagnostic journey, with the findings revealing how a lack of awareness among clinicians of the PCD symptom pattern can lead to a delayed diagnosis. Parents discussed the emotional and practical impact of a PCD diagnosis and the coping strategies employed to deal with challenges arising following a diagnosis. Parents use a variety of different lifestyle changes to accommodate their child's treatment regimen and to cope with disruptive life events such as the COVID-19 pandemic. This study provides valuable insights into parental adjustment and adaptation to a PCD diagnosis and management regimen. Going forward, this research highlights the need for integrated social care for PCD patients and their families.

COVID-19 symptom surveillance in immunocompromised children and young people in the UK: a prospective observational cohort study.

OBJECTIVES: To describe the frequency of symptoms compatible with SARS-CoV-2 infection in immunocompromised children and young people in the UK during the SARS-CoV-2 pandemic. To describe patient/parent anxiety regarding SARS-CoV-2 infection in this cohort. DESIGN: A prospective observational cohort study. SETTING: 46 centres across the UK between 16 March and 4 July 2020. A weekly online questionnaire based on the International Severe Acute Respiratory and emerging Infections Consortium-WHO Case Report Form was used to collect participant reported data on symptoms, test results, National Health Service attendance, hospital admission and impact on daily life. PARTICIPANTS: 1490 immunocompromised children, defined as those requiring an annual influenza vaccination due to their underlying condition or medication. MAIN OUTCOME MEASURES: Incidence of SARS-CoV-2-like symptoms and patient/parent anxiety score. RESULTS: Over 16 weeks during the first wave of the pandemic, no SARS-CoV-2 infection was diagnosed in this large immunocompromised paediatric cohort (median age 11 years, 54.4% female). 110 symptomatic participants underwent a test for SARS-CoV-2; all were negative. 922 (67.4%) participants reported at least one symptom consistent with suspected SARS-CoV-2 infection over the study period. 476 (34.8%) reported three or more symptoms. The most frequently reported symptoms included joint pain, fatigue, headache, nausea and muscle pain. SARS-CoV-2 testing during this period was performed on admitted patients only. 137 participants had their medication suspended or changed during the study period due to assumed COVID-19 disease risk. 62% reported high levels of anxiety (scores of 7-10 out of 10) at the start of the study, with anxiety levels remaining high throughout the study period. CONCLUSIONS: Although symptoms related to SARS-CoV-2 infection in children were common, there were no positive tests in this large immunocompromised cohort. Symptom-based screening to facilitate early detection of SARS-CoV-2 infection may not be helpful in these individuals. Patient/parent anxiety about SARS-CoV-2 infection was high. TRIAL REGISTRATION NUMBER: NCT04382508.

Mental health service users' perceptions of data sharing and data protection: a qualitative report.

BACKGROUND: To improve health services, social, economic and health data should be shared and linked to create a full narrative of lived experience. Mental health data sharing is often considered a particularly sensitive area. OBJECTIVE: To assess mental health service users' perceptions regarding the current practice of administrative data-driven research. METHOD: We conducted a focus group using case study scenarios. Themes and subthemes were analysed using qualitative methods. RESULTS: Participants were generally happy for data owners to share their health, social and economic data if the purpose was transparent and if the information would inform and improve health policy and practice. Participants were less keen on sharing data through digital applications. CONCLUSION: This case study informs a data linkage study protocol. Research teams and database owners should strive to educate service users on data protection and create dissent opportunities.

Age at onset, childhood psychopathology, and 2-year outcome in psychotic bipolar disorder.

OBJECTIVE: The relationships of age at onset and childhood psychopathology to 2-year clinical and functional outcomes in first-admission patients with bipolar I disorder were examined. METHOD: Patients with bipolar I disorder (N=123) presenting with psychotic symptoms were followed over a 2-year period. Age at onset was stratified into <19 and >or=19 years. Childhood psychopathology was categorized as behavior problems, other psychopathology, and none. Functional and clinical outcomes were rated with standard measures. RESULTS: Childhood psychopathology and age at onset were independently related to poorer functional and clinical outcome. In the multivariate models that included psychopathology, age at onset, sex, and education, early age at onset was related to incomplete remission, and childhood psychopathology was related to functional outcome. CONCLUSIONS: Childhood psychopathology and age at onset contribute independently to outcomes of bipolar disorder. Childhood psychopathology is a much stronger predictor of functioning than age at onset.