Mouse models of NADK2 deficiency analyzed for metabolic and gene expression changes to elucidate pathophysiology.

NADK2 encodes the mitochondrial form of nicotinamide adenine dinucleotide (NAD) kinase, which phosphorylates NAD. Rare recessive mutations in human NADK2 are associated with a syndromic neurological mitochondrial disease that includes metabolic changes, such as hyperlysinemia and 2,4 dienoyl CoA reductase (DECR) deficiency. However, the full pathophysiology resulting from NADK2 deficiency is not known. Here, we describe two chemically induced mouse mutations in Nadk2-S326L and S330P-which cause severe neuromuscular disease and shorten lifespan. The S330P allele was characterized in detail and shown to have marked denervation of neuromuscular junctions by 5 weeks of age and muscle atrophy by 11 weeks of age. Cerebellar Purkinje cells also showed progressive degeneration in this model. Transcriptome profiling on brain and muscle was performed at early and late disease stages. In addition, metabolomic profiling was performed on the brain, muscle, liver and spinal cord at the same ages and on plasma at 5 weeks. Combined transcriptomic and metabolomic analyses identified hyperlysinemia, DECR deficiency and generalized metabolic dysfunction in Nadk2 mutant mice, indicating relevance to the human disease. We compared findings from the Nadk model to equivalent RNA sequencing and metabolomic datasets from a mouse model of infantile neuroaxonal dystrophy, caused by recessive mutations in Pla2g6. This enabled us to identify disrupted biological processes that are common between these mouse models of neurological disease, as well as those processes that are gene-specific. These findings improve our understanding of the pathophysiology of neuromuscular diseases and describe mouse models that will be useful for future preclinical studies.

PRELIMINARY IMPACT OF SUPPORTIVE HOUSING ON HOSPITAL UTILIZATION FOR INDIVIDUALS EXPERIENCING HOMELESSNESS.

Housing instability has been shown to negatively impact physical and mental health, with a corresponding increase in health care utilization. In 2019, through a Maryland Medicaid 1115 Health Choice Waiver, 10 Baltimore city hospitals joined with the city of Baltimore and the local nonprofit Health Care for the Homeless to support an innovative program that provides permanent housing and wraparound services to individuals at risk of homelessness. Here, we describe the inception of the program and its subsequent expansion with the investment of the city hospitals. Participants in the program experienced a 48% reduction in all hospital visits and a 51% reduction in emergency department visits in the 12 months following their receipt of housing compared to the 12 months before enrollment. These data suggest the potential health benefits of housing and supportive services as an intervention.

Inclusive and intersectoral: community health improvement planning opportunities to advance the social determinants of health and health equity.

BACKGROUND: Community health improvement plans (CHIPs) are strategic planning tools that help local communities identify and address their public health needs. Many local health departments have developed a CHIP, yet there is a lack of research on the extent to which these plans address root causes of health disparities such as the social determinants of health. This study aims to inventory the social determinants of health included in 13 CHIPs and examine facilitators and challenges faced by local health departments and partners when trying to include the social determinants of health. METHODS: We conducted a comparative plan evaluation by scoring 13 CHIPs on their inclusion of equity orientation, inclusive planning processes, and five social determinants of health: health care access and quality, the neighborhood and built environment, economic stability, social and community context, and education access and quality. To supplement the plan evaluation, we conducted 32 in-depth interviews with CHIP leaders and stakeholders to understand the factors contributing to the inclusion and exclusion of the social determinants of health in the planning process. RESULTS: CHIPs received an average score of 49/100 for the inclusion of the social determinants of health. Most plans addressed health care access and quality and the neighborhood and built environment, but they often did not address economic stability, the social and community context, and education access and quality. Regarding their overall equity orientation, CHIPs received an average score of 35/100, reflecting a relative lack of attention to equity and inclusive planning processes in the plans. Interviews revealed that challenges engaging partners, making clear connections between CHIPs and social determinants, and a lack of capacity or public and partner support often led to the exclusion of the social determinants of health. Recommendations to improve planning processes include improving data infrastructure, providing resources for dedicated planning staff and community engagement incentives, and centering equity throughout the planning process. CONCLUSIONS: Although local health departments can leverage CHIPs to improve population health and address health disparities, they face a range of challenges to including the social determinants of health in CHIPs. Additional resourcing and improved data are needed to facilitate broader inclusion of these determinants, and more work is needed to elevate equity throughout these planning processes.

Mental Health and Mental Health Care Utilization Across Political Affiliation in U.S. Adults.

In the past decade, depression has become more visible in the public conversation; depression has also become bound in national divides. We sought to assess (1) whether positive screen for depression is associated with political party affiliation and (2) whether use of mental health care varies by political affiliation. Positive screen for depression did not differ significantly for Republicans versus Democrats in Spring 2023. However, Republicans were less likely to have received treatment for their mental health in the past 12 months. Depression is a bipartisan issue, and improving access to mental health care could be a priority that unites political groups during the upcoming election season.

Two new mouse models of Gjb1-associated Charcot-Marie-Tooth disease type 1X.

BACKGROUND: Charcot-Marie-Tooth disease type 1X is caused by mutations in GJB1, which is the second most common gene associated with inherited peripheral neuropathy. The GJB1 gene encodes connexin 32 (CX32), a gap junction protein expressed in myelinating glial cells. The gene is X-linked, and the mutations cause a loss of function. AIMS: A large number of disease-associated variants have been identified, and many result in mistrafficking and mislocalization of the protein. An existing knockout mouse lacking Gjb1 expression provides a valid animal model of CMT1X, but the complete lack of protein may not fully recapitulate the disease mechanisms caused by aberrant CX32 proteins. To better represent the spectrum of human CMT1X-associated mutations, we have generated a new Gjb1 knockin mouse model. METHODS: CRISPR/Cas9 genome editing was used to produce mice carrying the R15Q mutation in Gjb1. In addition, we identified a second allele with an early frame shift mutation in codon 7 (del2). Mice were analyzed using clinically relevant molecular, histological, neurophysiological, and behavioral assays. RESULTS: Both alleles produce protein detectable by immunofluorescence in Schwann cells, with some protein properly localizing to nodes of Ranvier. However, both alleles also result in peripheral neuropathy with thinly myelinated and demyelinated axons, as well as degenerating and regenerating axons, predominantly in distal motor nerves. Nerve conduction velocities were only mildly reduced at later ages and compound muscle action potential amplitudes were not reduced. Levels of neurofilament light chain in plasma were elevated in both alleles. The del2 mice have an onset at ~3 months of age, whereas the R15Q mice had a later onset at 5-6 months of age, suggesting a milder loss of function. Both alleles performed comparably to wild type littermates in accelerating rotarod and grip strength tests of neuromuscular performance. INTERPRETATION: We have generated and characterized two new mouse models of CMT1X that will be useful for future mechanistic and preclinical studies.

Pathways to social well-being of children with intellectual disability: testing the Family Investment Model.

BACKGROUND: Social well-being, including prosocial and peer relationship skills, independence and co-operation, is a particularly important developmental outcome in intellectual disability (ID). The present study investigated pathways to social well-being through the early years' family environment, particularly the role of parental investments in mediating the path from family poverty to child social well-being. METHODS: In line with the Family Investment Model (FIM), we tested whether parental investments between 3 and 5 years of age mediate the impact of family poverty at 9 months of age on children's social well-being at 7 years. Structural equation models were fitted to data from 555 children with ID identified from a UK population-based cohort. RESULTS: Findings indicated that home learning investments and the structural home environment (though not play) significantly mediated the effect of family poverty on children's social skills, albeit in different directions. While all parental investments reduced in the presence of poverty, the home learning environment appeared to promote social well-being, whereas the structural home environment did not. Sensitivity analyses controlling for co-occurring autism confirmed the pattern of findings. Child gender, ethnicity and parental educational qualifications did not moderate the mediational relationships, suggesting that FIM pathways to social well-being were relevant to all families. CONCLUSIONS: The FIM provides a helpful framework to map developmental pathways for children with an ID. Parental investments related to home learning, the structural home environment and play are reduced in the presence of poverty although their impact on child social well-being appears to differ.

Predictors and moderators of the response of adults with intellectual disabilities and depression to behavioural activation and guided self-help therapies.

BACKGROUND: No previous studies have reported predictors and moderators of outcome of psychological therapies for depression experienced by adults with intellectual disabilities (IDs). We investigated baseline variables as outcome predictors and moderators based on a randomised controlled trial where behavioural activation was compared with guided self-help. METHODS: This study was an exploratory secondary data analysis of data collected during a randomised clinical trial. Participants (n = 161) were randomised to behavioural activation or guided self-help and followed up for 12 months. Pre-treatment variables were included if they have previously been shown to be associated with an increased risk of having depression in adults with IDs or have been reported as a potential predictor or moderator of outcome of treatment for depression with psychological therapies. The primary outcome measure, the Glasgow Depression Scale for Adults with Learning Disabilities (GDS-LD), was used as the dependant variable in mixed effects regression analyses testing for predictors and moderators of outcome, with baseline GDS-LD, treatment group, study centre and antidepressant use as fixed effects, and therapist as a random effect. RESULTS: Higher baseline anxiety (mean difference in outcome associated with a 1 point increase in anxiety 0.164, 95% confidence interval [CI] 0.031, 0.297; P = 0.016), lower performance intelligence quotient (IQ) (mean difference in outcome associated with a 1 point increase in IQ 0.145, 95% CI 0.009, 0.280; P = 0.037) and hearing impairment (mean difference 3.449, 95% CI 0.466, 6.432; P = 0.024) were predictors of poorer outcomes, whilst greater severity of depressive symptoms at baseline (mean difference in outcome associated with 1 point increase in depression -0.160, 95% CI -0.806, -0.414; P < 0.001), higher expectation of change (mean difference in outcome associated with a 1 point increase in expectation of change -1.013, 95% CI -1.711, -0.314; p 0.005) and greater percentage of therapy sessions attended (mean difference in outcome with 1 point increase in percentage of sessions attended -0.058, 95% CI -0.099, -0.016; P = 0.007) were predictors of more positive outcomes for treatment after adjusting for randomised group allocation. The final model included severity of depressive and anxiety symptoms, lower WASI performance IQ subscale, hearing impairment, higher expectation of change and percentage of therapy sessions attended and explained 35.3% of the variance in the total GDS-LD score at 12 months (R2  = 0.353, F4, 128  = 17.24, P < 0.001). There is no evidence that baseline variables had a moderating effect on outcome for treatment with behavioural activation or guided self-help. CONCLUSIONS: Our results suggest that baseline variables may be useful predictors of outcomes of psychological therapies for adults with IDs. Further research is required to examine the value of these potential predictors. However, our findings suggest that therapists consider how baseline variables may enable them to tailor their therapeutic approach when using psychological therapies to treat depression experienced by adults with IDs.

Public support for U.S. social safety net policies throughout the COVID-19 pandemic.

COVID-19 has stretched the U.S. social safety net and prompted federal legislation designed to ameliorate the pandemic's health and economic impacts. We surveyed a nationally representative cohort of 1222 U.S. adults in April 2020 and November 2020 to evaluate changes in public opinion about 11 social safety net policies and the role of government over the course of the pandemic. A majority of U.S. adults supported six policies at both time points, including policies guaranteeing two weeks of paid sick leave; enacting universal health insurance; increasing the federal minimum wage; and increasing government spending on construction projects, business tax credits, and employment education and training. From April to November 2020, public support was stable for nine of the 11 policies but declined nearly 10 percentage points for policies guaranteeing two weeks paid sick leave (from 76% support in April 2020 to 67% support in November 2020) and extending unemployment insurance benefits (51% to 42%). Declines in support for these two policies were concentrated among those with higher incomes, more education, in better health status, the employed, and those with health insurance. The share of respondents believing in a strong role of government also declined from 33% in April to 26% in November 2020 (p > 0.05). Despite these shifts, we observed consistent majority support for several policies enacted during the pandemic, including guaranteeing paid sick leave and business tax credits, as well as employment-related policies.

The prevalence of potential drug-drug interactions in adults with intellectual disability.

BACKGROUND: There is a high use of medications in adults with intellectual disability (ID). One implication of taking multiple medications is the potential for drug-drug interactions (DDIs). However, despite this being well highlighted in the mainstream literature, little is known about the incidence or associations of DDIs in the ID population. METHODS: This study describes the prevalence, patterns and associations of potential DDIs in a total administrative sample of adults with ID known to services in Jersey. Demographic, health-related and medication data were collected from 217 adults known to ID services. Data were collected using a face-to-face survey. The Anatomical Therapeutic Chemical classification system was used to categorise medications, and Stockley's Drug Interaction Checker was used to classify potential DDIs. Drug-drug pairings were considered to be of clinical significance if they were to be 'avoided, adjusted, monitored or required further information'. RESULTS: Potential DDIs of clinical significance were common. Exposure to potential DDIs of clinical significance was associated with being female, taking more than five medications (polypharmacy), living in residential care and having more health conditions. A simple regression was used to understand the effect of number of prescribed medications on potential DDIs of clinical significance. Every prescribed drug led to a 0.87 (95% confidence interval: 0.72-1.00) increase in having a potential DDI of clinical significance. CONCLUSION: Adults with ID who live in residential care, who are female, exposed to polypharmacy and have more health conditions may be more likely to have potential DDIs of clinical significance. Urgent consideration needs to be given to the potential of DDIs in this population given their exposure to high levels of medication.

The impact of the COVID-19 pandemic on the health of adults with intellectual impairment: evidence from two longitudinal UK surveys.

BACKGROUND: People with an intellectual impairment experience high levels of social and health inequalities. We investigated the impact of COVID-19 on the physical and mental health of people with intellectual impairment, controlling for demographic risk, socio-economic circumstances and pre-pandemic health levels. METHOD: Data were drawn from two UK birth cohorts that surveyed their participants on the impact of COVID-19 in May 2020: the Millennium Cohort Study (20-year-old participants) and the British Cohort Survey (50-year-old participants). Health outcomes (COVID-19 infection, COVID-19 symptoms, self-reported physical health, mental health, health service use and impact on health behaviours) were compared between people with and without intellectual impairment, adjusting for gender and ethnicity. Differences were further adjusted for self-reported health pre-pandemic and the impact of COVID-19 on socio-economic circumstances. RESULTS: Controlling for gender and ethnicity, poor health was reported less often by younger adults [relative risks (RR): 0.44 95% confidence interval (CI) 0.23, 0.86] and more often by older adults (RR: 1.99 95% CI 1.45, 2.73) with intellectual impairment compared with peers. Older adults were also more likely to experience fever and loss of taste/smell. Adjusting for pre-pandemic health and socio-economic circumstances eliminated some differences in the older cohort, but not in the younger one. CONCLUSION: In young adulthood, the impact of COVID-19 on health outcomes was not negative. The pattern was reversed in later adulthood, although differences were mostly eliminated after adjustment suggesting a socio-economic and age gradient of COVID-19 impacts on intellectual impairment.

The willingness of UK adults with intellectual disabilities to take COVID-19 vaccines.

BACKGROUND: Given the much greater COVID-19 mortality risk experienced by people with intellectual disabilities (ID), understanding the willingness of people with ID to take a COVID-19 vaccine is a major public health issue. METHOD: In December 2020 to February 2021, across the United Kingdom, 621 adults with ID were interviewed remotely and 348 family carers or support workers of adults with ID with greater needs completed an online survey, including a question on willingness to take a COVID-19 vaccine if offered. RESULTS: Eighty-seven per cent of interviewees with ID were willing to take a COVID-19 vaccine, with willingness associated with white ethnicity, having already had a flu vaccine, gaining information about COVID-19 from television but not from social media, and knowing COVID-19 social restrictions rules. A percentage of 81.7% of surveyed carers of adults with ID with greater needs reported that the person would be willing to take a COVID-19 vaccine, with willingness associated with white ethnicity, having a health condition of concern in the context of COVID-19, having had a flu vaccine, being close to someone who had died due to COVID-19, and having shielded at some point during the pandemic. CONCLUSIONS: Reported willingness to take the COVID-19 vaccine is high among adults with ID in the United Kingdom, with factors associated with willingness having clear implications for public health policy and practice.

Vaccine hesitancy among working-age adults with/without disability in the UK.

OBJECTIVES: To estimate levels of COVID-19 vaccine hesitancy among working-age adults with disabilities in the United Kingdom. STUDY DESIGN: Cross-sectional survey. METHODS: Secondary analysis of data collected on a nationally representative sample of 10,114 respondents aged 16-64 years. RESULTS: The adjusted relative risk for hesitancy among respondents with a disability was 0.92 (95% CI 0.67-1.27). There were stronger associations between gender and hesitancy and ethnic status and hesitancy among participants with a disability. The most common reasons cited by people with disabilities who were hesitant were: concern about the future effects of the vaccine, not trusting vaccines and concern about the side effects of vaccination. CONCLUSIONS: The higher rates of vaccine hesitancy among women with disabilities and among people from minority ethnic groups with disabilities are concerning.

Polypharmacy and psychotropic polypharmacy in adults with intellectual disability: a cross-sectional total population study.

BACKGROUND: Adults with intellectual disability (ID) are prescribed high levels of medication, with polypharmacy and psychotropic polypharmacy common. However, reported rates vary between studies, and there has been an over-reliance on obtaining data from convenience samples. The objective of this study was to determine the prevalence of medication use and polypharmacy in a population-level sample of adults with IDs. Factors associated with polypharmacy and psychotropic polypharmacy are explored. METHODS: We used a total population sample of 217 adults with IDs known to services in Jersey (sampling frame n = 285). The Anatomical Therapeutic Chemical classification system was used to categorise medications that participants were currently taking on a regular basis. We examined associations of polypharmacy and psychotropic polypharmacy with socio-economic status, health and demographic variables using univariate and multivariate analyses. RESULTS: A total of 83.4% of participants were prescribed medication, with high doses common. Of the participants, 38.2% were exposed to polypharmacy while 23% of participants were exposed to psychotropic polypharmacy. After controlling for demographic, health and socio-economic characteristics, polypharmacy was significantly associated with older age, increased severity of ID, living in a residential setting and having increased comorbidities. Psychotropic polypharmacy was associated with being male, being aged 50+ years and having had a psychiatric diagnosis over the life course. Being prescribed psychotropic drugs above the defined daily dose was not associated with having had a psychiatric diagnosis over the life course, suggesting the possibility of 'off label' prescribing. CONCLUSIONS: Our results indicate that medication use, in high doses, alongside polypharmacy and psychotropic polypharmacy are highly prevalent in adults with ID. The exposure to multiple medications increases the risk of developing adverse drug events, drug-drug interactions and medication-related problems. Future population-level, prospective cohort studies should examine the prevalence of polypharmacy and psychotropic polypharmacy using standardised definitions and consider the potential impact of adverse drug events, drug-drug interactions and medication-related problems in this population.

Impact of intended and relative dose intensity of R-CHOP in a large, consecutive cohort of elderly diffuse large B-cell lymphoma patients treated with curative intent: no difference in cumulative incidence of relapse comparing patients by age.

BACKGROUND: The increasing incidence of diffuse large B-cell lymphoma (DLBCL) in ageing populations places a significant burden on healthcare systems. Co-morbidity, frailty, and reduced organ and physiological reserve contribute to treatment-related complications. The optimal dose intensity of R-CHOP to optimize outcome across different ages with variable frailty and comorbidity burden is unclear. OBJECTIVES AND METHODS: We examined the influence of intended (IDI) and relative (RDI) dose intensity of the combination of cyclophosphamide and doxorubicin, age and comorbidity on outcomes for DLBCL patients ≥70 years in a representative, consecutive cohort across eight UK centres (2009-2018). We determined predictors of survival using multivariable Cox regression, and predictors of recurrence before death using competing risks regression. RESULTS: Porgression-free survival (PFS) and overall survival (OS) were significantly inferior in patients ≥80 vs. 70-79 years (P < 0.001). In contrast, 2-year cumulative relapse incidence, when accounting for non-relapse mortality as a competing risk, was no different between 70-79 vs. ≥80 years (P = 0.27) or comorbidity status (CIRS-G: 0-6 vs. >6) (P = 0.27). In 70-79 years, patients with an IDI ≥80% had a significantly improved PFS and OS (P < 0.001) compared to IDI < 80%. Conversely, in patients ≥80 years, there was no difference in PFS (P = 0.88) or OS (P = 0.75) according to IDI <80% vs. ≥80%. On multivariable analysis, when comparing by age, there was a significantly higher cumulative relapse rate for patients aged 70-79 years with an IDI <80% (vs. >80%) (P = 0.04) but not for patients ≥80 years comparing IDI (P = 0.32). CONCLUSION: 'R-mini-CHOP' provides adequate lymphoma-specific disease control and represents a reasonable treatment option in elderly patients ≥80 years aiming for cure.

Risk of exposure to air pollution among British children with and without intellectual disabilities.

BACKGROUND: Exposure to outdoor air pollution is a well-established risk factor for a range of adverse health conditions. No previous study has quantified the extent to which children with intellectual disability (ID) may be exposed to outdoor air pollution. METHODS: Secondary analysis of data extracted from the UK's Millennium Cohort Study, a nationally representative sample of over 18 000 UK children born 2000-2002. RESULTS: Averaging across ages, children with IDs were 33% more likely to live in areas with high levels of diesel particulate matter, 30% more likely to live in areas with high levels of nitrogen dioxide, 30% more likely to live in areas with high levels of carbon monoxide and 17% more likely to live in areas with high levels of sulphur dioxide. CONCLUSIONS: Levels of exposure to outdoor air pollution among children with ID are significantly higher than those of families of children without ID. Exposure to outdoor air pollution may be one of the pathways that contributes to the health inequities experienced by people with IDs.

Total chemical synthesis of lassomycin and lassomycin-amide.

Herein we report a practical synthetic route to the lasso peptide lassomycin () and C-terminal variant lassomycin-amide (). The biological evaluation of peptides and against Mycobacterium tuberculosis revealed that neither had any activity against this bacterium. This lack of biological activity has led us to propose that naturally occurring lassomycin may actually exhibit a standard lasso peptide threaded conformation rather than the previously reported unthreaded structure.

Predictors of self-reported alcohol use and attitudes toward alcohol among 11-year-old British children with and without intellectual disability.

BACKGROUND: Reducing harmful levels of alcohol consumption among children is an important public health concern internationally and in many high income countries. Little is known about levels and predictors of alcohol use among children with intellectual disability (ID). METHOD: Secondary analysis of child self-report data at age 11 years collected in the UK's Millennium Cohort Study. RESULTS: Children with ID were significantly more likely to: have used alcohol in the last 4 weeks; to have had five or more alcoholic drinks on one occasion; to have had five or more alcoholic drinks or been intoxicated on one occasion; to have more positive attitudes about the psychological and social benefits of drinking; and to have less negative attitudes about the social and physical costs of drinking. Potentially harmful levels of drinking (intoxication or 5+ alcoholic drinks on one occasion) among children with ID were associated with child smoking, having friends who use alcohol, reporting that drinking makes it easier to make friends, and reporting that drinking reduces worrying. Children with ID accounted for 9% of all children with potentially harmful levels of drinking. CONCLUSION: Public health interventions to reduce potentially harmful drinking among children in general must recognise that children with ID are a potentially high risk group and ensure that interventions are appropriately adjusted to take account of their particular needs and situation. Future research in this area is needed to untangle the causal pathways between attitudes toward alcohol and alcohol use among children with ID and the extent to which levels of alcohol use and predictors of alcohol use may be moderated by severity of ID.

Exposure to second hand tobacco smoke at home and child smoking at age 11 among British children with and without intellectual disability.

BACKGROUND: The exposure of children to second hand tobacco smoke (SHS) is a well-established risk factor for a range of adverse health conditions in childhood and later life. Little is known about the extent to which children with intellectual disability (ID) may be exposed to SHS. Our aim in this study was to estimate the risk of childhood exposure to SHS and early experience of smoking among children with and without ID in a nationally representative cohort of British children. METHOD: Secondary analysis of data extracted from the UK's Millennium Cohort Study, a nationally representative sample of over 18,000 UK children born 2000-2002. RESULTS: Children with ID are significantly more likely than their peers to be exposed to SHS and to have themselves experimented with smoking by age 11. Controlling for between-group differences in socio-economic position eliminated the increased risk of exposure to SHS and significantly attenuated, but did not eliminate, increased risk of experimenting with smoking by age 11. CONCLUSIONS: Levels of exposure to SHS among children with ID are typical of those of families of children without ID living in similar socio-economic circumstances. The results lend no support to the hypothesis that increased rates of parental smoking may be associated with any additional 'burden of care' experienced by parents of children with ID. Nevertheless, it will be important to ensure that evidence-based interventions to reduce exposure to SHS are tailored to the specific needs of families supporting children with ID (e.g. through the provision of disability-friendly child care arrangements).

The health of parents with and without intellectual impairment in the UK.

BACKGROUND: Little is known about the health and well-being of the 'hidden majority' of parents with mild intellectual disability (ID), who are less likely to be in contact with disability services. METHOD: We sought to add to knowledge in this area by examining the health and living conditions of parents with and without intellectual impairment in a large contemporary nationally representative sample of UK parents aged between 16 and 49 years old (n = 14 371). RESULTS: Our results indicated that, as expected, parents with intellectual impairment were at significantly greater risk than other parents of having poorer self-reported general, mental and physical health. They were also at significantly greater risk of experiencing higher rates of household socio-economic disadvantage and environmental adversities and lower rates of neighbourhood social capital and intergenerational support. Adjusting risk estimates to take account of between group differences in household socio-economic disadvantage eliminated statistically significant differences in health status between parents with and without intellectual impairment on all but one indicator (obesity). Further adjusting risk estimates to take account of between group differences in neighbourhood adversity, neighbourhood social capital and intergenerational support had minimal impact on the results. CONCLUSIONS: That controlling for between-group differences in exposure to socio-economic disadvantage largely eliminated evidence of poorer health among parents with intellectual impairment is consistent with the view that a significant proportion of the poorer health of people with IDs may be attributable to their poorer living conditions rather than biological factors associated with ID per se.

A preliminary controlled trial of a trans-diagnostic programme for cognitive behaviour therapy with adults with intellectual disability.

BACKGROUND: Several studies have found a heightened prevalence of mental health disorders in people with intellectual disabilities (ID). There have been a number of successful case series and two promising controlled treatment trials of cognitive behaviour therapy (CBT) for emotional disorders (excluding anger) for people with ID. Several authors have promoted the development of trans-diagnostic approaches to cognitive treatment. The present study extends this work with the development and evaluation of a trans-diagnostic treatment manual for CBT in people with ID. METHOD: A controlled treatment trial was conducted with 12 participants in treatment and waiting list control data. Each treatment participant was matched to a control on age, IQ, presenting problem, and Brief Symptom Inventory (BSI) global severity index (GSI) score. The treatment group was also evaluated on the Glasgow anxiety and depression scales and was followed up for 3 to 6 months after treatment. RESULTS: There were no significant differences between groups at baseline. Following treatment, the CBT group was significantly improved when compared with the control group on the GSI scale of the BSI. The ancovas for all other measures were not significant but there were significant improvements for the treatment group on all scaled except BSI depression from pre to post-CBT. Gains were maintained to follow up, and changes were associated with large effect sizes. CONCLUSIONS: It was possible to treat a range of symptoms and psychiatric diagnoses with a general trans-diagnostic CBT manual. The effects of therapy were promising, suggesting that the participants could respond to treatment in a meaningful and helpful manner and supporting the case for further evaluation of the trans-diagnostic approach in ID.