title: Developing better information about the health and health care of people with developmental disabilities in Scotland
Creators: name: Hughes-McCormack, Laura Anne
Publisher: name: University of Glasgow
Year: 2021

description: Introduction: Neurodevelopmental disorders are a group of disorders that manifest early in development, and include intellectual disabilities and autism, among others. Intellectual disabilities refer to impairments in intellectual functioning (an intelligence quotient &lt;70), together with deficits in adaptive functioning (need for support for daily personal independence and social functioning), with onset during the developmental period. Autism is characterised by persistent deficits in social communication and social interactions across multiple contexts, and restricted, repetitive patterns of behaviour, interests or activities, with onset of these symptoms in the early developmental period. People with intellectual disabilities and people with autism are thought to experience high levels of physical and mental health problems and earlier mortality than other people on average. Yet, there is a dearth of empirical evidence about the health of people with intellectual disabilities and people with autism which presents a barrier to understanding the complex factors that produce differential health outcomes. Ensuring adults with intellectual disabilities or autism live not only longer but healthier lives is a priority for the World Health Organisation and the Scottish Government. Methods: The portfolio of publications (n=15) presented in this mixed methods PhD thesis, represents a selection of my peer reviewed publications in international scientific journals since 2015. I [Laura Hughes-McCormack] am the lead author on 4 of these publications and co-author on 11 publications (including being second author on 8 of these) which were prepared from research I undertook within the Scottish Learning Disabilities Observatory (SLDO) at the Institute of Health and Wellbeing, University of Glasgow. This research programme was funded by the Scottish Government in 2014 to provide evidence on the health of people with intellectual disabilities and autism in Scotland, and thus to inform the development of public policy. The research presented includes systematic reviews, quantitative research, data linkage research, and is presented in three themes throughout this thesis, as follows. Theme I. Health of people with intellectual disabilities and autism; seven of the nine quantitative studies presented under this theme/section analysed data from the Scottish Census, relating to people with intellectual disabilities or autism from 94% of the Scottish population (n=5,269,054) who responded to the Census in 2011. Two further studies (a quantitative study and a systematic review study) are reported, which investigated sedentary behaviour and oral health. Theme II. Health care of people with intellectual disabilities and Down syndrome; to quantify the management of long-term conditions, data for a population-based cohort of people with intellectual disabilities (n=721) was compared using an established evidence-based approach to measuring the quality of primary health care for all people without intellectual disabilities (n=764,672) in the largest health board in Scotland, throughout 2007-2010. A further systematic review study investigated hospital admissions for physical health conditions in adults with intellectual disabilities. Theme III. Survival/ death of people with intellectual disabilities and Down syndrome; two systematic reviews are reported, investigating deaths in people with intellectual disabilities and people with Down syndrome, and a further data linkage study which investigated birth and death rates and hospitalisations (throughout a 25-year period) among children/young people with Down syndrome in Scotland. Each theme includes a clear overview of the background/ rationale, methodology, results and impact of this body of work in relation to the development of better information on health and health care of people with intellectual disabilities and autism in Scotland. Results: Theme I: Health of people with intellectual disabilities and autism: Findings of the Scottish Census 2011 studies (I-VII), show that poor health was more common for people with intellectual disabilities (odds of 43 in statistically predicting poor general health) and they were seven times more likely to report a current mental health condition than people without intellectual disabilities. Autism had odds of 11.3 in predicting poor general health in children and young people, and odds of 7.5 in adults. Comorbidities were found to be common among people with autism. Other studies presented under this theme (VIII, IX) show adults with intellectual disabilities have higher levels, and different correlates, of sedentary behaviour and poorer oral health. Theme II: Health care of people with intellectual disabilities and Down syndrome: Findings from two studies (X, XI) show, people with intellectual disabilities were receiving lower quality health care compared to other people across all long‐term conditions investigated on 38/57 (66.7%) quality indicators. A follow up study, comparing baseline data to data in 2014 found adults with intellectual disabilities still had a lower proportion of indicators met than the general population; but by 2014, the healthcare inequality gap had reduced compared with 2007-10. A systematic review (XII), further investigating the quality of health care of people with intellectual disabilities, found people with intellectual disabilities were admitted to hospital more frequently than the general population for physical conditions which if managed effectively at the primary care level, should not lead to hospital admission, although evidence is limited. Theme III: Survival/ death of people with intellectual disabilities and Down syndrome: Findings from the two systematic reviews (XIII, XIV) found mortality rates are higher in (1) the intellectual disabilities population and (2) the Down syndrome population, compared with the general population. For the intellectual disabilities’ population, an average age of death of 20 years lower was found compared to the general population. For the Down syndrome population, compared with the general population, an average age of death of 28 years lower was found although survival rates have improved over time. Patterns of cause of death were different for people with intellectual disabilities and people with Down syndrome compared to the general population. The data linkage study (XV) found the incidence of Down syndrome live-births was 1.0/1,000 births over the last 25 years. More children and young people with Down syndrome died (n=92; 7.4%) over the 25-year period compared to controls (n=23; 0.4%); that is 18.5 times more. There was increased risk of hospitalisation as more of the Down syndrome group had at least one admission (1,105 [89.5%] versus 3,305 [53.5%]; adjusted HR=1.84 [1.68, 2.01]). Re-admissions, emergency admissions and length of stay were also higher for the Down syndrome group. Conclusions: This thesis has presented for each of the three inter-related themes, a range of my peer reviewed publications from international journals. A previous dearth of empirical evidence about the health and health care of people with intellectual disabilities and people with autism has presented barriers to understanding the complex factors that produce differential health outcomes. The research presented in this thesis has provided robust evidence on the extent of poor health, health care, and premature mortality among people with intellectual disabilities, and people with autism which has not previously been quantified in research. This evidence has led to shaping Scottish policy and practice to support the needs of people with intellectual disabilities, for example, the most recent learning disabilities strategy in Scotland, The Keys to Life, updated in 2019, includes input from my research.