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Amenable deaths among adults with intellectual and developmental disabilities including Down syndrome: An Ontario population‐based cohort study
Abstract
Background:
Rates of death and avoidable deaths are reportedly higher among people with intellectual and developmental disabilities. This study contributes to our understanding of how mortality and intellectual and development disabilities are associated.
Method:
General population and intellectual and developmental disabilities adult cohorts were defined using linked administrative data. All-cause and amenable deaths between 2010 and 2015 were reported for these cohorts and subcohorts with and without Down syndrome. Cox proportional hazards models evaluated the impact of potential contributors to amenable deaths.
Results:
Adults with intellectual and developmental disabilities had higher all-cause (6.1 vs. 1.6%) and amenable death percentages (21.4 vs. 14.1%) than general population comparators. Within intellectual and developmental disabilities, those with Down syndrome had higher all-cause (12.0 vs. 6.0%) but lower amenable death percentages (19.2 vs. 21.8%) than those without.
Conclusions:
Results suggest that interventions to reduce amenable deaths target provider-care-recipient interactions and coordination across care and support sectors.
... One of them reported a higher all-cause (6.1% vs 1.6%) and amenable (21.4% vs 14.1%) mortality levels compared with the general population, but rates for all avoidable mortality were not provided. 12 The other study reported 1 year age-standardised mortality rates for years 2011-2014 to be between 30.3 and 37.4 for Manitoba adults with intellectual and developmental disabilities compared with the matched comparison group, meaning that 30.3-37.4 times more deaths occurred in this population than would be expected to occur in the Ontario population. 13 A further study on adults with intellectual and developmental disabilities from Manitoba, Canada reported crude avoidable premature mortality rates per 1000 person-years to be between 2.3 and 3.3 for years 2013-2015, meaning that avoidable premature mortality was 2.3-3.3 times more prevalent among Manitoba adults with intellectual and developmental disabilities compared with the matched comparison group. ...
Background
Studies on avoidable mortality in adults with intellectual disabilities are limited, as are studies on causes of death.
Objectives
We aimed to quantify mortality rates, and causes, and identify factors (i.e., age, sex, Scottish Index of Multiple Deprivation (SIMD)) related to avoidable mortality in adults with intellectual disabilities.
Design
A record linkage national cohort study.
Setting
A cohort of adults with intellectual disabilities with or without co-occurring autism, aged 25+ years and a randomly selected comparison group aged 25+ years without intellectual disabilities or autism identified from Scotland’s Census, 2011. Census records were linked to the National Records of Scotland Statutory Register of Deaths database to ascertain all deaths from 2011 to 2019.
Participants
We analysed data on 14 477 adults with intellectual disabilities aged 25+ years and a randomly selected comparison group of 506 207 adults aged 25+ without intellectual disabilities identified from Scotland’s Census 2011.
Primary and secondary outcome measures
We ran χ ² tests and t-tests to investigate individual characteristics and differences in age at death for adults with intellectual disabilities compared with peers in the general population. Cox proportional hazard models were fitted to calculate risk of mortality (all-cause, avoidable, treatable, preventable) unadjusted and adjusted for age, sex and SIMD. We then calculated mortality rates, using crude and indirect standardisation methods.
Results
During the 8.5-year follow-up, 23.7% (crude death rate of 3033.3 per 100 000) of adults with intellectual disabilities died compared with 13.8% of controls. The median age at death among adults aged 25+ with intellectual disabilities was 65.0 years compared with 80.0 years for adults without intellectual disabilities. For all-cause mortality, the age-standardised mortality ratio (SMR) in the population with intellectual disabilities was 3.1 (95% CI 3.0 to 3.2). The SMRs were higher for the youngest age groups, women and in the most affluent areas. This was also the case for SMRs for avoidable, treatable and preventable deaths. For the population of adults with intellectual disabilities, 31.7% of recorded deaths were considered avoidable, 21.1% were treatable and 19.9% were preventable. In the controls, 18.2% of deaths were considered avoidable, 8.8% treatable and 14.7% preventable. Down syndrome and dementia were the two most commonly recorded underlying causes of death for people with intellectual disabilities while malignant neoplasm of bronchus and lung and acute myocardial infarction were most commonly recorded in the general population.
Conclusions
Risk of all-cause, avoidable, treatable and preventable mortality was higher for adults with intellectual disabilities than their peers. The highest SMRs were observed for youngest adults, women and individuals living in the most affluent areas.
... 3 4 Evidence from across the globe shows that people with intellectual disability are more likely to die premature deaths than their counterparts without intellectual disability, with many of these being potentially avoidable with the provision of good quality healthcare. [5][6][7][8] Improving care for people with an intellectual disability and reducing health inequalities experienced by this group is a priority for governments and healthcare systems. 9 Adults with intellectual disability are more likely than those without to be admitted to hospital and their admissions are longer, 10 11 yet they frequently report poor experiences of hospital care. ...
Objective
Intellectual disability liaison nurses in general hospitals could enhance access to high-quality, adapted healthcare and improve outcomes. We aimed to explore associations between the input of intellectual disability liaison nurses and the quality of care in people with intellectual disability who are admitted to hospital.
Design
Retrospective analysis of a national dataset of mortality reviews.
Setting
General hospitals in England.
Participants
4742 adults with intellectual disability who died in hospital between 2016 and 2021 and whose deaths were reviewed as part of the Learning from Lives and Deaths mortality review programme.
Outcome measures
We used logistic regression to compare the sociodemographic and clinical characteristics of those who did, and did not, receive input from an intellectual disability liaison nurse. We explored associations between liaison nurse input, care processes and overall quality of care.
Results
One-third of people with intellectual disability who died in hospital in England between 2016 and 2021 had input from an intellectual disability liaison nurse. Intellectual disability liaison nurse input was not evenly distributed across England and was more common in those who died of cancer. Having an intellectual disability liaison nurse involved in an individual’s care was associated with increased likelihood of reasonable adjustments being made to care (adjusted OR (aOR) 1.95, 95% CI 1.63 to 2.32) and of best practice being identified (aOR 1.37, 95% CI 1.17 to 1.60) but was not associated with a rating of overall quality of care received (aOR 0.94, 95% CI 0.78 to 1.12).
Conclusions
Intellectual disability liaison nurses see only a minority of people with intellectual disability who are admitted to hospital in England. Increasing the availability of intellectual disability liaison nurses could improve care for this disadvantaged group.
... Many physical and mental health conditions are more common in people with intellectual disabilities than in the general population such as epilepsy, cerebral palsy, attention deficit hyperactivity disorder and anxiety disorders (Buckley et al., 2020;Hughes-McCormack et al., 2017;Liao et al., 2021), with high rates of polypharmacy (Haider et al., 2014;O'Dwyer et al., 2016). Median age of death has been estimated at 62 years (White et al., 2021) and avoidable and premature death is a significant international issue (Cooper et al., 2020;Doyle et al., 2021;Lin et al., 2023;Tyrer et al., 2021). ...
Background
People with intellectual disabilities may experience frailty earlier than the general population. This scoping review aimed to investigate how frailty is defined, assessed, and managed in adults with an intellectual disability; factors associated with frailty; and the potential impact of COVID-19 on frailty identification and management.
Method
Databases were searched from January 2016 to July 2023 for studies that investigated frailty in individuals with intellectual disabilities.
Results
Twenty studies met the inclusion criteria. Frailty prevalence varied between 9% and 84%. Greater severity of intellectual disability, presence of Down syndrome, older age, polypharmacy, and group home living were associated with frailty. Multiagency working, trusted relationships and provision of evidence-based information may all be beneficial in frailty management.
Conclusion
Frailty is common for people with intellectual disabilities and is best identified with measures specifically designed for this population. Future research should evaluate interventions to manage frailty and improve lives.
... Therefore, customized treatment and healthcare strategies are needed according to the disability severity and type. [14][15][16] However, previous studies have mainly focused on people with specific types of disabilities such as intellectual, physical disability, and people aged 65 and older. Because of the barriers to accessing healthcare and a high risk of severe cardiovascular diseases, such as stroke, in younger individuals with disabilities, the incidence and long-term trends of stroke should be evaluated in the entire population with disabilities. ...
Background
This study investigated 10-year trend in the incidence and prevalence of ischemic, hemorrhagic, and overall strokes according to the severity and type of disability between people with and without disabilities.
Methods
This serial cross-sectional analysis was conducted using national health information data during a 10-year period from 2008 to 2017. Age-standardized incidence and prevalence were analyzed for each year, according to the presence, severity, and type of disability. The odds ratio (OR) of stroke was examined using multivariable logistic regression after adjusting for socio-demographic and clinical variables collected in 2017.
Findings
In total, 413,398,084 people were enrolled between 2008 and 2017. In 2017, 43,552,192 people aged 19 or older were included and 5.8% was disabled. For 10 years, age-standardized incidence of ischemic and hemorrhagic stroke decreased significantly regardless of the presence of disability. However, age-standardized incidence of stroke in disabled were almost 2.5 times higher than the non-disabled in 2017. Stroke occurs 20 years earlier in people with disabilities than in those without disabilities. In 2017, people with disabilities had higher odds of stroke compared to those without disability (OR = 4.11, 95% confidence interval [CI]: 4.06–4.16), particularly among those with severe disabilities (OR = 4.75, 95% CI: 4.67–4.84). People with major internal organ impairment showed the highest incidence of stroke (OR = 5.95, 95% CI: 5.73–6.17). The main risk factors for stroke presented in this study were disability factors, chronic diseases, and advanced age.
Interpretation
People with disabilities are at a greater risk of developing stroke incidence. Developing a public health policy and identifying the risk factors for stroke in people with disabilities would be beneficial.
Funding
This work was supported by a 10.13039/501100003725National Research Foundation of Korea (NRF) grant funded by the 10.13039/100009950Ministry of Education (No. 2022R1I1A3070074).
Aim
To describe how specialised models of registered nurse‐led care and support can play a vital role in the health and quality of life of someone with intellectual disability and multiple chronic and complex health problems.
Design
Single instrumental case study.
Methods
Data collection occurred between March and August 2023, and included interviews with parents, historical case files, descriptions of the nurse‐led model of care and a list of the daily registered nurse‐led interventions.
Results
The evidence presented strongly support the positive impact a nurse‐led model of care can have for persons with profound disability and complex health conditions, and their families. The improved quality of life observed in this project underscores the significant role nurses play in enhancing the well‐being of this vulnerable population.
Conclusion
Further research on a larger scale should be completed to create a robust foundation of a specialised, nurse‐led model of care for individuals with profound disability and complex health conditions.
Implications
There is room for funded intermediate models of care as the health system cannot be expected, and nor is it appropriate, to provide ongoing care for all people with intellectual disability.
Reporting Method
This study adhered to the COREQ guidelines for qualitative research.
Patient or Public Contribution
No patient or public contribution.
Background
We now have sufficient evidence demonstrating inequalities in specific avoidable causes of death for adults with intellectual disability compared to their peers without intellectual disability. Apart from covid‐19, the largest differentials that disadvantage people with intellectual disability are in relation to pneumonia, aspiration pneumonia, epilepsy, cerebrovascular disease, ischaemic heart disease, deep vein thrombosis, diabetes and sepsis. The aim of this systematic review is to report on strategies at the individual, population or policy levels aimed at preventing these conditions that are applicable to adults with intellectual disability and that have been based on or recommended by research.
Methods
Systematic review of PUBMED, EMBASE, CINAHL, PsychInfo, Social Care Online, International Bibliography of the Social Sciences (IBSS), Web of Science, Scopus, Overton, the Cochrane Library and Google Scholar databases was carried out. Searches were completed on 30 June 2023. Quantitative, qualitative and mixed‐methods research; systematic, scoping or evidence‐based reviews; and audit and reports of mortality reviews were included. Publications included in the review were about preventing the eight potentially avoidable causes of death.
Findings
Ninety‐four papers were included in the review (9 in relation to pneumonia; 11 for aspiration pneumonia; 18 for sudden unexpected death in epilepsy; 7 for cerebrovascular disease; 8 for ischaemic heart disease; 4 for deep vein thrombosis; 31 for diabetes; 6 for sepsis). The eight most frequently occurring potentially avoidable causes of death in people with intellectual disability are very different medical conditions, but they shared striking similarities in how they could be prevented. The literature overwhelmingly implicated the need to make lifestyle changes to address obesity, lack of exercise and poor nutrition, and to have regular medical reviews. In addition, ‘whole‐population’ approaches are required that look beyond the individual to the social determinants of health.
Conclusions
We found little peer‐reviewed evidence specifically about preventing these conditions in people with intellectual disability. However, most of the literature about preventative strategies pertaining to the general population was applicable to people with intellectual disability, albeit that some ‘reasonable adjustments’ would be required.
Importance
Knowledge of the health challenges and mortality in people with intellectual disability (ID) should guide health policies and practices in contemporary society.
Objective
To examine premature mortality in individuals with ID.
Design, Setting, and Participants
This population-based longitudinal cohort study obtained data from several national health care, education, and population registers in Sweden. Two registers were used to identify individuals with ID: the National Patient Register and the Halmstad University Register on Pupils With Intellectual Disability. Two cohorts were created: cohort 1 comprised young adults (born between 1980 and 1991) with mild ID, and cohort 2 comprised individuals (born between 1932 and 2013) with mild ID or moderate to profound ID; each cohort had matched reference cohorts. Data analyses were conducted between June 1, 2020, and March 31, 2021.
Exposures
Mild or moderate to profound ID.
Main Outcomes and Measures
The primary outcome was overall (all-cause) mortality, and the secondary outcomes were cause-specific mortality and potentially avoidable mortality.
Results
Cohort 1 included 13 541 young adults with mild ID (mean [SD] age at death, 24.53 [3.66] years; 7826 men [57.8%]), and its matched reference cohort consisted of 135 410 individuals. Cohort 2 included 24 059 individuals with mild ID (mean [SD] age at death, 52.01 [16.88] years; 13 649 male individuals [56.7%]) and 26 602 individuals with moderate to profound ID (mean [SD] age at death, 42.16 [21.68] years; 15 338 male individuals [57.7%]); its matched reference cohorts consisted of 240 590 individuals with mild ID and 266 020 with moderate to profound ID. Young adults with mild ID had increased overall mortality risk compared with the matched reference cohort (odds ratio [OR], 2.86; 95% CI, 2.33-3.50), specifically excess mortality in neoplasms (OR, 3.58; 95% CI, 2.02-6.35), diseases of the nervous system (OR, 40.00; 95% CI, 18.43-86.80) and circulatory system (OR, 9.24; 95% CI, 4.76-17.95). Among deaths that were amenable to health care (OR, 7.75; 95% CI, 4.85-12.39), 55% were attributed to epilepsy. In cohort 2, increased risk of overall mortality was observed among both individuals with mild ID (OR, 6.21; 95% CI, 5.79-6.66) and moderate to profound ID (OR, 13.15; 95% CI, 12.52-13.81) compared with the matched reference cohorts. Those with moderate to profound ID had a higher risk in several cause-of-death categories compared with those with mild ID or the matched reference cohort. Adjustment for epilepsy and congenital malformations attenuated the associations. The relative risk of premature death was higher in women (OR, 6.23; 95% CI, 4.42-8.79) than in men (OR, 1.99; 95% CI, 1.53-2.60), but the absolute risk of mortality was similar (0.9% for women vs 0.9% for men).
Conclusions and Relevance
This study found excess premature mortality and high risk of deaths with causes that were potentially amenable to health care intervention among people with ID. This finding suggests that this patient population faces persistent health challenges and inequality in health care encounters.
In England, the national mortality review programme for people with intellectual disabilities, the LeDeR programme, was established in 2015. The programme supports local areas to review the deaths of all people with intellectual disabilities aged 4 years and over. Each death has an initial review; if indicated, a full multi-agency review takes place. The learning from the mortality reviews contributes to service improvements locally and nationally. This paper describes the programme’s introduction and processes, exploring the challenges faced, and the successes achieved. It considers the background and rationale for the programme and the steps taken during its implementation, in order that others can learn from our experiences. Now the programme is established, its focus needs to shift so that we have a better understanding about how the findings of mortality reviews are leading to local and national service improvements and their impact.
Objectives
To investigate mortality rates and causes in children and young people with intellectual disabilities.
Design
Retrospective cohort; individual record linkage between Scotland’s annual pupil census and National Records of Scotland death register.
Setting
General community.
Participants
Pupils receiving local authority-funded schooling in Scotland, 2008 to 2013, with an Additional Support Need due to intellectual disabilities, compared with other pupils.
Main outcome measures
Deaths up to 2015: age of death, age-standardised mortality ratios (age-SMRs); causes of death including cause-specific age-SMRs; avoidable deaths as defined by the UK Office of National Statistics.
Results
18 278/947 922 (1.9%) pupils had intellectual disabilities. 106 died over 67 342 person-years (crude mortality rate=157/100 000 person-years), compared with 458 controls over 3 672 224 person-years (crude mortality rate=12/100 000 person-years). Age-SMR was 11.6 (95% CI 9.6 to 14.0); 16.6 (95% CI 12.2 to 22.6) for female pupils and 9.8 (95% CI 7.7 to 12.5) for male pupils. Most common main underlying causes were diseases of the nervous system, followed by congenital anomalies; most common all-contributing causes were diseases of the nervous system, followed by respiratory system; most common specific contributing causes were cerebral palsy, pneumonia, respiratory failure and epilepsy. For all contributing causes, SMR was 98.8 (95% CI 69.9 to 139.7) for congenital anomalies, 76.5 (95% CI 58.9 to 99.4) for nervous system, 63.7 (95% CI 37.0 to 109.7) for digestive system, 55.3 (95% CI 42.5 to 72.1) for respiratory system, 32.1 (95% CI 17.8 to 57.9) for endocrine and 14.8 (95% CI 8.9 to 24.5) for circulatory system. External causes accounted for 46% of control deaths, but the SMR for external-related deaths was still higher (3.6 (95% CI 2.2 to 5.8)) for pupils with intellectual disabilities. Deaths amenable to good care were common.
Conclusion
Pupils with intellectual disabilities were much more likely to die than their peers, and had a different pattern of causes, including amenable deaths across a wide range of disease categories. Improvements are needed to reduce amenable deaths, for example, epilepsy-related and dysphagia, and to support families of children with life-limiting conditions.
Objectives
The goals of this study were to examine and compare (a) the annual adult mortality rates and (b) the most commonly reported underlying causes of death between a cohort of Manitobans with intellectual and developmental disabilities (IDD) and a matched comparison group without IDD.
Methods
Using linked health and nonhealth administrative data, a cohort of Manitoba adults with IDD, aged 25–99 years in 2012, was identified. Each person in the study cohort was matched with three persons without IDD based on age, sex, region of residence, and morbidity level. The two groups were followed for three years (2013–2015). Crude annual adult mortality rates and avoidable premature mortality rates were calculated. The leading causes of death over the 3‐year study period were tabulated by ICD‐10 chapter. Coding of the underlying causes of death was reviewed.
Results
The crude annual mortality rates for Manitoba adults with IDD were 1.8–2.4 times higher than those for the matched comparison group and remained stable over time. Disparities in mortality rates for the IDD cohort relative to the matched comparison group decreased with increasing age. No significant sex differences were found. The leading causes of death among the IDD cohort were diseases of the circulatory system, cancer, and diseases of the respiratory system. Avoidable premature deaths were 2.3–3.3 times more prevalent among Manitoba adults with IDD compared to the matched comparison group. An IDD diagnostic code was reported as cause of death in 2.11% of cases.
Conclusions
The excess mortality among adults with IDD should be monitored in Manitoba and all other jurisdictions and attention paid to the causes of death and their coding.
Objectives
To investigate mortality in adults with intellectual disabilities: rates, causes, place, demographic and clinical predictors.
Design
Cohort study with record linkage to death data.
Setting
General community.
Participants
961/1023 (94%) adults (16–83 years; mean=44.1 years; 54.6% male) with intellectual disabilities, clinically examined in 2001–2004; subsequently record-linked to their National Health Service number, allowing linkage to death certificate data, 2018.
Outcome measures
Standardised mortality ratios (SMRs), underlying and all contributing causes of death, avoidable deaths, place, and demographic and clinical predictors of death.
Results
294/961 (30.6%) had died; 64/179 (35.8%) with Down syndrome, 230/783 (29.4%) without Down syndrome. SMR overall=2.24 (1.98, 2.49); Down syndrome adults=5.28 (3.98, 6.57), adults without Down syndrome=1.93 (1.68, 2.18); male=1.69 (1.42, 1.95), female=3.48 (2.90, 4.06). SMRs decreased as age increased. More severe intellectual disabilities increased SMR, but ability was not retained in the multivariable model. SMRs were higher for most International Statistical Classification of Diseases and Related Health Problems, 10th Revision chapters. For adults without Down syndrome, aspiration/reflux/choking and respiratory infection were the the most common underlying causes of mortality; for Down syndrome adults ‘Down syndrome’, and dementia were most common. Amenable deaths (29.8%) were double that in the general population (14%); 60.3% died in hospital. Mortality risk related to percutaneous endoscopic gastrostomy/tube fed, Down syndrome, diabetes, lower respiratory tract infection at cohort-entry, smoking, epilepsy, hearing impairment, increasing number of prescribed drugs, increasing age. Bowel incontinence reduced mortality risk.
Conclusions
Adults with intellectual disabilities with and without Down syndrome have different SMRs and causes of death which should be separately reported. Both die younger, from different causes than other people. Some mortality risks are similar to other people, with earlier mortality reflecting more multimorbidity; amenable deaths are also common. This should inform actions to reduce early mortality, for example, training to avoid aspiration/choking, pain identification to address problems before they are advanced, and reasonable adjustments to improve healthcare quality.
Objective: To determine whether coding a developmental disability as the underlying cause of death obscures mortality trends of adults with developmental disability.
Design: National Vital Statistics System 2012–2016 US Multiple Cause-of-Death Mortality files.
Setting: USA.
Participants: Adults with a developmental disability indicated on their death certificate aged 18 through 103 at the time of death. The study population included 33 154 adults who died between 1 January 2012 and 31 December 2016.
Primary outcome and measures: Decedents with a developmental disability coded as the underlying cause of death on the death certificate were identified using the International Statistical Classification of Diseases and Related Health Problems, Tenth Revision code for intellectual disability, cerebral palsy, Down syndrome or other developmental disability. Death certificates that coded a developmental disability as the underlying cause
of death were revised using a sequential underlying cause of death revision process.
Results: There were 33 154 decedents with developmental disability: 7901 with intellectual disability, 11 895 with cerebral palsy, 9114 with Down syndrome, 2479 with
other developmental disabilities and 1765 with multiple developmental disabilities. Among all decedents, 48.5% had a developmental disability coded as the underlying
cause of death, obscuring higher rates of choking deaths among all decedents and dementia and Alzheimer’s disease among decedents with Down syndrome.
Conclusion: Death certificates that recorded the developmental disability in Part I of the death certificate were more likely to code disability as the underlying cause of death. While revising these death certificates provides a short-term corrective to mortality trends for this population, the severity and extent of this problem warrants a long-term change involving more precise instructions to record developmental disabilities only in Part II of the death certificate.
Background
Mortality studies can help reduce health inequalities by informing public policy through a better understanding of causes of death and comorbidities. Mortality studies often rely on Medical Certificates of Cause of Death (MCCD) for data.
Method
A systematic review was undertaken to identify the extent and nature of issues in recording causes of death for people with intellectual disability on MCCD.
Results
Fifteen of the 25 articles included in the literature review raised concerns about the accuracy of MCCD in identifying the cause(s) of death of people with intellectual disability. The most frequent issues were the under‐reporting of intellectual disability on MCCD, and listing intellectual disability or an associated condition as an underlying cause of death.
Conclusions
Concerns about the accuracy and reliability of MCCD for people with intellectual disability raise questions about mortality data based on MCCD. Clear guidance is required from WHO for those completing MCCD for people with intellectual disability.
Access to necessary supports and services is extremely important for the wellbeing and, ultimately, quality of life (QOL) of family caregivers. By understanding patterns of health service utilization, we can better tailor current services to meet caregivers’ needs. Framed by Andersen’s Behavioral Model, predisposing, enabling and need factors associated with service utilization, and their relation to QOL, were examined in a cross-sectional sample of 156 Australian caregivers of children and adolescents with autism spectrum disorder (ASD). Participants completed an online survey to assess services accessed, QOL (Quality of Life in Autism -
Part A), health beliefs (Health Beliefs Questionnaire), ASD symptom severity (Quality of Life in Autism - Part B), and caregiver distress (Depression Anxiety Stress Scales – 21 item). Enabling (i.e. income) and need variables (i.e. no. of children with ASD, parental distress) were significantly (p < 0.05) associated with service use and QOL. This was confirmed by multivariate regression analyses: family structure (i.e. number of children with
ASD; OR = 7.22, CI: 1.77–29.54, p < .01) and caregiver distress (OR = 1.04, CI: 1.0–1.07, p < .05) were identified as unique contributors to service access. Both of these variables also helped to explain a sizeable portion of the variance in QOL scores (R2 = .32, adj. R2 = .24, p <.001). Personal barriers to health care utilization exist for parents of children with ASD which impact their decision to access help. There is, however, a need for further research to expand Andersen’s model by examining subjective and objective QOL indicators applicable to the caregiver cohort.
Background
Sweden has closed all institutions and imposed legislation to ensure service and support for individuals with intellectual disability (ID). Understanding mortality among older individuals with ID is essential to inform development of health promotion and disease control strategies. We investigated patterns and risk of mortality among older adults with ID in Sweden.
Methods
This retrospective cohort study compared older adults aged 55 years and older with ID with a control population. Participants were followed during 2002–2015 or death, and censored if they moved out of Sweden. Individuals with ID were identified from two national registers: one covering all specialist health-care visits (out-patient visits and hospitalisation) and the other covering people accessing social/support services. Individuals with ID (n = 15,289) were matched with a control population by sex, birth year, and year of first hospitalisation/out-patient visit/access to LSS services. Cause-of-death data were recorded using International Classification of Diseases, Tenth Revision. Cox proportional hazards regression were conducted to assess if overall and cause-specific mortality rate among individuals with ID was higher than in the Swedish population.
Results
The overall mortality rate among individuals with ID was 2483 per 100,000 people compared with 810 in the control population. Among those who died, more individuals with ID were younger than 75 years and unmarried. Leading causes of death among individuals with ID were circulatory diseases (34%), respiratory diseases (17%) and neoplasms (15%). Leading causes of death in a sub-sample with Down syndrome (DS) were respiratory diseases (37%), circulatory diseases (26%) and mental/behavioural disorders (11%). Epilepsy and pneumonitis were more common among individuals with ID than controls. Alzheimer’s disease was common in the control population and individuals with DS, but not among those with ID when DS was excluded. Individuals with ID had a higher overall mortality risk (hazard ratio [HR] 4.1, 95% confidence interval [CI] 4.0–4.3) and respiratory disease death risk (HR 12.5, 95% CI 10.9–14.2) than controls.
Conclusion
Older adults with ID in Sweden carry a higher mortality risk compared with the general population, mainly attributable to respiratory, nervous and circulatory diseases. Care for this group, particularly during the terminal stage of illness, needs to be tailored based on understanding of their main health problem.
Electronic supplementary material
The online version of this article (10.1186/s12877-017-0665-3) contains supplementary material, which is available to authorized users.
Background
Immigrants have been shown to possess a health advantage, yet are also more likely to reside in arduous economic conditions. Little is known about if and how the socioeconomic gradient for all-cause, premature and avoidable mortality differs according to immigration status.
Methods
Using several linked population-based vital and demographic databases from Ontario, we examined a cohort of all deaths in the province between 2002 and 2012. We constructed count models, adjusted for relevant covariates, to attain age-adjusted mortality rates and rate ratios for all-cause, premature and avoidable mortality across income quintile in immigrants and long-term residents, stratified by sex.
Results
A downward gradient in age-adjusted all-cause mortality was observed with increasing income quintile, in immigrants (males: Q5: 13.32, Q1: 20.18; females: Q5: 9.88, Q1: 12.51) and long-term residents (males: Q5: 33.25, Q1: 57.67; females: Q5: 22.31, Q1: 36.76). Comparing the lowest and highest income quintiles, male and female immigrants had a 56% and 28% lower all-cause mortality rate, respectively. Similar trends were observed for premature and avoidable mortality. Although immigrants had consistently lower mortality rates compared with long-term residents, trends only differed statistically across immigration status for females (p<0.05).
Conclusions
This study illustrated the presence of income disparities as it pertains to all-cause, premature, and avoidable mortality, irrespective of immigration status. Additionally, the immigrant health advantage was observed and income disparities were less pronounced in immigrants compared with long-term residents. These findings support the need to examine the factors that drive inequalities in mortality within and across immigration status.
Objectives
To investigate mortality and its causes in adults over the age of 20 years with intellectual disability (ID).
Design, setting and participants
Retrospective population-based standardised mortality of the ID and Comparison cohorts. The ID cohort comprised 42 204 individuals who registered for disability services with ID as a primary or secondary diagnosis from 2005 to 2011 in New South Wales (NSW). The Comparison cohort was obtained from published deaths in NSW from the Australian Bureau of Statistics (ABS) from 2005 to 2011.
Main outcome measures
We measured and compared Age Standardised Mortality Rate (ASMR), Comparative Mortality Figure (CMF), years of productive life lost (YPLL) and proportion of deaths with potentially avoidable causes in an ID cohort with an NSW general population cohort.
Results
There were 19 362 adults in the ID cohort which experienced 732 (4%) deaths at a median age of 54 years. Age Standardised Mortality Rates increased with age for both cohorts. Overall comparative mortality figure was 1.3, but was substantially higher for the 20–44 (4.0) and 45–64 (2.3) age groups. YPLL was 137/1000 people in the ID cohort and 49 in the comparison cohort. Cause of death in ID cohort was dominated by respiratory, circulatory, neoplasm and nervous system. After recoding deaths previously attributed to the aetiology of the disability, 38% of deaths in the ID cohort and 17% in the comparison cohort were potentially avoidable.
Conclusions
Adults with ID experience premature mortality and over-representation of potentially avoidable deaths. A national system of reporting of deaths in adults with ID is required. Inclusion in health policy and services development and in health promotion programmes is urgently required to address premature deaths and health inequalities for adults with ID.
Background:
We aimed to ascertain the average age at death (AD) in the intellectual disability population for each gender and compare them to those of the general population during 1970-2012.
Methods:
By analysing medical records, we calculated the ADs of all deceased clients (N = 1236) of two district organizations responsible for intellectual disability services. Statistics Finland's database generated data regarding ADs of all inhabitants who had died after having resided in same district.
Results:
During the follow-up, average ADs for the intellectual disability population and general population increased, and simultaneously the AD difference between these populations decreased. In the 2000s, the AD difference between the intellectual disability population and the whole population was 22 years for men (95% CI: -24 to -20) and 30 years for women (95% CI: -33 to -27). In 2000s, the mean AD of those with mild-to-moderate intellectual disability (IQ 50-69) for women and men was 56 (SD17) and 54 (SD18), and those with severe to profound intellectual disability (IQ<50), 44 (SD23) and 43 (SD21).
Conclusions:
Intellectual disability is still a considerable risk factor for early death. Among the intellectual disability population, unlike in general population, the lifespans of women and men are equal.
Objectives:
To describe mortality among adults with intellectual disability in England in comparison with the general population.
Methods:
We conducted a cohort study from 2009 to 2013 using data from 343 general practices. Adults with intellectual disability (n = 16 666; 656 deaths) were compared with age-, gender-, and practice-matched controls (n = 113 562; 1358 deaths).
Results:
Adults with intellectual disability had higher mortality rates than controls (hazard ratio [HR] = 3.6; 95% confidence interval [CI] = 3.3, 3.9). This risk remained high after adjustment for comorbidity, smoking, and deprivation (HR = 3.1; 95% CI = 2.7, 3.4); it was even higher among adults with intellectual disability and Down syndrome or epilepsy. A total of 37.0% of all deaths among adults with intellectual disability were classified as being amenable to health care intervention, compared with 22.5% in the general population (HR = 5.9; 95% CI = 5.1, 6.8).
Conclusions:
Mortality among adults with intellectual disability is markedly elevated in comparison with the general population, with more than a third of deaths potentially amenable to health care interventions. This mortality disparity suggests the need to improve access to, and quality of, health care among people with intellectual disability. (Am J Public Health. Published online ahead of print June 16, 2016: e1-e8. doi:10.2105/AJPH.2016.303240).
Objectives:
This study aimed at ascertaining the standardized mortality ratios (SMR) for those with an intellectual disability (ID) in Finland.
Materials and methods:
We used the statistical database of the national insurance institution of Finland and Statistics Finland's mean population figures. We determined the number of individuals who received benefits (disability allowance, disability pension, or care allowance for pensioners) due to an ID diagnosis and the number of those whose benefit had been terminated due to death during the years 1996-2011.
Results:
SMR for females with a mild ID (IQ 50-69) was 2.8 (95% CI: 2.60-3.01) and for males 2.0 (95% CI: 1.88-2.14), and for females with a severe ID (IQ <50) 5.2 (95% CI: 4.99-5.50) and for males 2.6 (95% CI: 2.48-2.72).
Conclusion:
This significant difference in the SMR figures between males and females with ID warrants further research.
Individuals with intellectual and developmental disabilities (IDD) experience high rates of social and health disadvantage. Planning effective services that meet the needs of this vulnerable population requires good population-based data that are collected on a routine, ongoing basis. However, in most jurisdictions, none of the commonly available data (e.g., health or disability benefits administrative data) completely captures the IDD population. To more accurately identify persons with IDD in a population, one solution is to link data across multiple sources. To do this, the authors report on an effort to create a linked database to identify a cohort of adults, aged 18–64, with IDD in Ontario and use these data to examine how the linkage can help study health and healthcare access. The linked dataset was created using four health and one disability income support databases. Standardized differences were used to compare sociodemographic and clinical characteristics of the IDD cohorts identified through the health, disability income support, and linked datasets. Indirect estimation was used to evaluate which IDD subgroups might be over- or underestimated if only a single source of data was available. The linked database identified a cohort of 66,484 adults with IDD (0.78% prevalence). The health and disability income support data each uniquely identified approximately a third of the cohort. Health data were more likely to identify younger adults (18–24 years), those with psychiatric illnesses, and hospitalized individuals. The disability income support data were more likely to identify adults aged 35–54 and those living in lower income neighborhoods. By linking multiple databases, the authors were able to identify a much larger cohort of individuals with IDD than if they had used a single data source. It also enabled the creation of a more accurate sociodemographic and clinical profile of this population as each source captured different segments of it.
The Confidential Inquiry into premature deaths of people with intellectual disabilities in England was commissioned to provide evidence about contributory factors to avoidable and premature deaths in this population.
The population-based Confidential Inquiry reviewed the deaths of people with intellectual disabilities aged 4 years and older who had been registered with a general practitioner in one of five Primary Care Trust areas of southwest England, who died between June 1, 2010, and May 31, 2012. A network of health, social-care, and voluntary-sector services; community contacts; and statutory agencies notified the Confidential Inquiry of all deaths of people with intellectual disabilities and provided core data. The Office for National Statistics provided data about the coding of individual cause of death certificates. Deaths were described as avoidable (preventable or amenable), according to Office for National Statistics definitions. Contributory factors to deaths were identified and quantified by the case investigator, verified by a local review panel meeting, and agreed by the Confidential Inquiry overview panel. Contributory factors were grouped into four domains: intrinsic to the individual, within the family and environment, care provision, and service provision. The deaths of a comparator group of people without intellectual disabilities but much the same in age, sex, and cause of death and registered at the same general practices as those with intellectual disabilities were also investigated.
The Confidential Inquiry reviewed the deaths of 247 people with intellectual disabilities. Nearly a quarter (22%, 54) of people with intellectual disabilities were younger than 50 years when they died, and the median age at death was 64 years (IQR 52-75). The median age at death of male individuals with intellectual disabilities was 65 years (IQR 54-76), 13 years younger than the median age at death of male individuals in the general population of England and Wales (78 years). The median age at death of female individuals with intellectual disabilities was 63 years (IQR 54-75), 20 years younger than the median age at death for female individuals in the general population (83 years). Avoidable deaths from causes amenable to change by good quality health care were more common in people with intellectual disabilities (37%, 90 of 244) than in the general population of England and Wales (13%). Contributory factors to premature deaths in a subset of people with intellectual disabilities compared with a comparator group of people without intellectual disabilities included problems in advanced care planning (p=0·0003), adherence to the Mental Capacity Act (p=0·0008), living in inappropriate accommodation (p<0·0001), adjusting care as needs changed (p=0·009), and carers not feeling listened to (p=0·006).
The Confidential Inquiry provides evidence of the substantial contribution of factors relating to the provision of care and health services to the health disparities between people with and without intellectual disabilities. It is imperative to examine care and service provision for this population as potentially contributory factors to their deaths-factors that can largely be ameliorated.
Department of Health for England.
Attention to disease and risk factor management is increasingly a feature of people with intellectual disability (ID) as an augmented life expectancy also exposes a growing number of age-related diseases. An additional concern is little attention to date to physical activity, nutrition, access to social support and other personal health choices and to environmental issues such as the impact of access to social support and the implications of individual's living arrangements. Method: Using a sample of 753 persons with ID from the intellectual disability supplement to the Irish longitudinal study on ageing (IDS-TILDA), forty three variables were grouped into environmental, predisposing, enabling , need and personal health choices clusters and hierarchical ordinary least squares regression examined the contribution of environmental, enabling, predisposing, need and all combinations of the sets of variables to personal health choices. Findings: Almost 32% of variance was explained primarily by need variables. Most significant relationships were with meeting up with family and friends (environmental), age, rating of health and worries about getting older (predis-posing), having public health insurance and nursing who come into the home (enabling) and presence of stroke, chronic constipation, functional limitations, high assistance needs with activities of daily living (need). Discussion: Taken together, the groupings of variables from the Anderson Model explained a modest amount of variance in the pursuit of positive personal health choices by people with ID. More work is clearly needed in developing evidence-based interventions and strategies, and in understanding the relationship between positive personal health choices of people with ID and health outcomes .
The purpose of this study was to validate autism spectrum disorder cases identified through claims-based case identification algorithms against a clinical review of medical charts. Charts were reviewed for 432 children who fell into one of the three following groups: (a) more than or equal to two claims with an autism spectrum disorder diagnosis code (n = 182), (b) one claim with an autism spectrum disorder diagnosis code (n = 190), and (c) those who had no claims for autism spectrum disorder but had claims for other developmental or neurological conditions (n = 60). The algorithm-based diagnoses were compared with documented autism spectrum disorders in the medical charts. The algorithm requiring more than or equal to two claims for autism spectrum disorder generated a positive predictive value of 87.4%, which suggests that such an algorithm is a valid means to identify true autism spectrum disorder cases in claims data.
Objective: This systematic review aims to assess the use and implementation of the Behavioral Model of Health Services Use developed by Ronald M. Andersen in recent studies explicity using this model.
Methods: A systematic search was conducted using PubMed in April 2011. The search strategy aimed to identify all articles in which the Andersen model had been applied and which had been published between 1998 and March 2011 in English or German. The search yielded a total of 328 articles. Two researchers independently reviewed the retrieved articles for possible inclusion using a three-step selection process (1. title/author, 2. abstract, 3. full text) with pre-defined inclusion and exclusion criteria for each step. 16 studies met all of the inclusion criteria and were used for analysis. A data extraction form was developed to collect information from articles on 17 categories including author, title, population description, aim of the study, methodological approach, use of the Andersen model, applied model version, and main results. The data collected were collated into six main categories and are presented accordingly.
Results: Andersen’s Behavioral Model (BM) has been used extensively in studies investigating the use of health services. The studies identified for this review showed that the model has been used in several areas of the health care system and in relation to very different diseases. The 1995 version of the BM was the version most frequently applied in the studies. However, the studies showed substantial differences in the variables used. The majority of the reviewed studies included age (N=15), marital status (N=13), gender/sex (N=12), education (N=11), and ethnicity (N=10) as predisposing factors and income/financial situation (N=10), health insurance (N=9), and having a usual source of care/family doctor (N=9) as enabling factors. As need factors, most of the studies included evaluated health status (N=13) and self-reported/perceived health (N=9) as well as a very wide variety of diseases. Although associations were found between the main factors examined in the studies and the utilization of health care, there was a lack of consistency in these findings. The context of the studies reviewed and the characteristics of the study populations seemed to have a strong impact on the existence, strength and direction of these associations.
Conclusions: Although the frequently used BM was explicitly employed as the theoretical background for the reviewed studies, their operationalizations of the model revealed that only a small common set of variables was used and that there were huge variations in the way these variables were categorized, especially as it concerns predisposing and enabling factors. This may stem from the secondary data sets used in the majority of the studies, which limited the variables available for study. Primary studies are urgently needed to enrich our understanding of health care utilization and the complexity of the processes shown in the BM.
Background
This study examines recent trends in the age‐at‐death disparity between adults with and without intellectual and/or developmental disabilities in the United States.
Method
Data were from the 2005–2017 U.S. death certificates. Average age at death was compared between adults whose death certificate did or did not report an intellectual and/or developmental disability.
Results
Age at death increased minimally for adults without, but markedly for adults with intellectual and/or developmental disabilities. As a result, the age‐at‐death disparity decreased: 2.2 years between adults with/without intellectual disability; 1.9 years between adults with/without Down syndrome; 2.7 years between adults with/without cerebral palsy; and 5.1 years between adults with/without rare developmental disabilities.
Conclusion
Evidence from this study demonstrates that the age‐at‐death disparity between adults who did or did not have an intellectual and/or developmental disability reported on their death certificate continues to decrease, but the magnitude of the remaining disparity varied considerably by type of disability.
Background
There is a need to systematically compare and contrast mortality predictors and disparities in people with intellectual disabilities (ID) for global prevention strategy development.
Method
Bibliographic databases and grey literature were searched using systematic review methodology and the machine learning tool “Abstrackr.”
Results
Fifty-four relevant articles and reports published from 2010 to 2019 were identified. Nearly all (n = 53) were from high-income countries. Mortality disparities were apparent and consistent across countries and publication years, with no evidence of a decrease over time. People with ID can still expect to live 12–23 years less than the general population and are particularly vulnerable to deaths from respiratory infections and epilepsy.
Conclusions
Both population and individual-level approaches to prevention are indicated to tackle the continuing mortality disparities in people with ID, including consideration of reasonable adjustments in general population efforts to reduce health inequalities.
We estimated deaths amenable to high-quality health care globally and then modeled the macroeconomic impact in low- and middle-income countries using two macroeconomic perspectives: a value-of-lost-output approach to project gross domestic product (GDP) losses annually for the period 2015-30, and a value-of-lost-welfare approach to estimate the present value of total economic welfare losses in 2015. We estimated that eight million amenable deaths occurred in 2015, 96 percent of them in low- and middle-income countries. The value of lost output resulted in a projected cumulative loss of 6.0 trillion in 2015. Inadequate access to high-quality health care results in significant mortality and imposes a macroeconomic burden that is inequitably distributed, with the largest relative burden falling on low-income countries. Given that these deaths are unnecessary and the projected GDP losses are avoidable, there is a strong ethical and economic case for promoting high-quality health care as an essential component of universal health coverage.
Background
It is thought that people with Down syndrome die younger than the general population, but that survival rates are improving.
Methods
Five databases were searched for keywords related to intellectual disabilities, Down syndrome and mortality. Strict inclusion criteria were applied. Information from 34 selected studies was tabulated, extracted and synthesized.
Results
People with Down syndrome died about 28 years younger than the general population. Congenital heart anomalies, comorbidities, low birthweight, and Black and minority ethnicity influenced earlier age of death, as did younger maternal age and poorer parental education. Congenital heart anomalies and respiratory conditions were the leading causes of death, and more common than in the general population. Survival rates have improved over time, particularly for those with congenital heart anomalies.
Conclusions
People with Down syndrome are living longer but still die younger of different causes than the general population. More robust comparative data are needed, and ethnic differences require further study.
Background:
Death of people with intellectual disabilities is considered to be earlier than for the general population.
Methods:
Databases were searched for key words on intellectual disabilities and death. Strict inclusion/exclusion criteria were used. Information was extracted from selected papers, tabulated and synthesized. Prospero registration number: CRD42015020161.
Results:
Of 19,111 retrieved articles, 27 met criteria. Death was earlier by 20 years. It has improved in recent decades; however, the same inequality gap with the general population remains. More severe intellectual disabilities, and/or additional comorbidities rendered it shortest. Standardized mortality rates showed a greater inequality for women than men. Respiratory disease and circulatory diseases (with greater congenital and lesser ischaemic disease compared with the general population) were the main causes of death. Cancer was less common, and cancer profile differed from the general population. Some deaths are potentially avoidable. All research is from high-income countries, and cause of death is surprisingly little investigated.
Conclusions:
Improved health care, including anticipatory care such as health checks, and initiatives addressing most relevant lifestyle behaviours and health risks are indicated.
Background:
People with intellectual disabilities (IDs) die at younger ages than the general population, but nationally representative and internationally comparable mortality data about people with ID, quantifying the extent and pattern of the excess, have not previously been reported for England.
Method:
We used data from the Clinical Practice Research Datalink database for April 2010 to March 2014 (CPRD GOLD September 2015). This source covered several hundred participating general practices comprising roughly 5% of the population of England in the period studied. General practitioner (GP) records identified people diagnosed by their GP as having ID. Linked national death certification data allowed us to derive corresponding mortality data for people with and without ID, overall and by cause.
Results:
Mortality rates for people with ID were significantly higher than for those without. Their all-cause standardised mortality ratio was 3.18. Their life expectancy at birth was 19.7 years lower than for people without ID. Circulatory and respiratory diseases and neoplasms were the three most common causes of death for them. Cerebrovascular disease, thrombophlebitis and pulmonary embolism all had standardised mortality ratios greater than 3 in people with ID. This has not been described before. Other potentially avoidable causes included epilepsy (3.9% of deaths), aspiration pneumonitis (3.6%) and colorectal cancer (2.4%). Avoidable mortality analysis showed a higher proportion of deaths from causes classified as amenable to good medical care but a lower proportion from preventable causes compared with people without ID. International comparison to areas for which data have been published in sufficient detail for calculation of directly standardised rates suggest England may have higher death rates for people with ID than areas in Canada and Finland, and lower death rates than Ireland or the State of Massachusetts in the USA.
Conclusions:
National data about mortality in people with ID provides a basis for public health interventions. Linked data using GP records to identify people with ID could provide comprehensive population-based monitoring in England, unbiased by the circumstances of illnesses or death; to date information governance constraints have prevented this. However, GPs in England currently identify only around 0.5% of the population as having ID, suggesting that individuals with mild, non-syndromic ID are largely missed. Notably common causes of death suggest control of cardiovascular risk factors, epilepsy and dysphagia, management of thrombotic risks and colorectal screening are important areas for health promotion initiatives.
Effect sizes are an important component of experimental design, data analysis and interpretation of statistical results. In some situations, an effect size of clinical or practical importance may be unknown to the researcher. In other situations, the researcher may be interested in comparing observed effect sizes to known standards to quantify clinical importance. In these cases, the notion of relative effect sizes (small, medium, large) can be useful as benchmarks. Although there is generally an extensive literature on relative effect sizes for continuous data, little of this research has focused on relative effect sizes for measures of risk that are common in epidemiological or biomedical studies. The aim of this paper, therefore, is to extend existing relative effect sizes to the relative risk, odds ratio, hazard ratio, rate ratio, and Mantel-Haenszel odds ratio for related samples. In most scenarios with equal group allocation, effect sizes of 1.22, 1.86 and 3.00 can be taken as small, medium and large respectively. The odds ratio for a non-rare event is a notable exception and modified relative effect sizes are 1.32, 2.38 and 4.70 in that situation.
Increases in the life expectancy of people with Intellectual Disability have followed similar trends to those found in the general population. With the exception of people with severe and multiple disabilities or Down syndrome, the life expectancy of this group now closely approximates with that of the general population. Middle and old age, which until 30 years ago were not recognized in this population, are now important parts of the life course of these individuals. Older adults with Intellectual Disabilities form a small, but significant and growing proportion of older people in the community. How these persons grow older and how symptoms and complications of the underlying cause of the Intellectual Disability will influence their life expectancy is of the utmost importance. © 2013 Wiley Periodicals, Inc. Dev Disabil Res Rev 2013;18:6-16.
Objective:
To compare the fidelity of administrative data with clinical data when researching Down syndrome (DS).
Methods:
From outpatient, inpatient, and emergency department administrative claims within our institution, we identified 252 patients aged 18-45 years with encounters coded for DS by the ICD9=758.0 from 2000 to 2008. We evaluated these cases for false-positive errors-cases in which DS was not actually present in clinical descriptions. Subsequently, we identified false-negative errors (cases in which DS was present without encounters coded as such) by examination of the medical records for all patients within our study frame who had one of several common DS comorbidities, including congenital heart disease, hypothyroidism, and atlantoaxial instability.
Results:
Among the 252 people with an administrative code for DS, 53 (21%) did not have DS documented in their medical record (false-positive error). While searching for false-negative errors, 29 additional patients were discovered with DS documented in the medical record who had not been previously identified. This led to a final cohort of 228 patients with DS. The presence of a billing code for DS had moderate sensitivity (87%) and positive predictive value (79%), but high specificity (99.9%).
Discussion:
Administrative claims misclassify a sizeable proportion of patients with DS. Judgments about quality of care on the basis of samples identified using administrative claims may not accurately reflect the experience of patients with the conditions in question. When using administrative databases to study the quality of care for patients with DS, diagnostic verification within the clinical record is advisable whenever possible.
Congenital heart disease (CHD) is common in patients with Down syndrome (DS), and these patients are living longer lives. The aim of this study was to describe the epidemiology of hospitalizations in adults with DS and CHD in the United States. Hospitalizations from 1998 to 2009 for adults aged 18 to 64 years with and without DS with CHD diagnoses associated with DS (atrioventricular canal defect, ventricular septal defect, tetralogy of Fallot, and patent ductus arteriosus) were analyzed using the Nationwide Inpatient Sample. Outcomes of interest were (1) in-hospital mortality, (2) common co-morbidities, (3) cardiac procedures, (4) hospital charges, and (5) length of stay. Multivariate modeling adjusted for age, gender, CHD diagnosis, and co-morbidities. There were 78,793 ± 2,653 CHD admissions, 9,088 ± 351 (11.5%) of which were associated with diagnoses of DS. The proportion of admissions associated with DS (DS/CHD) decreased from 15.2 ± 1.3% to 8.5 ± 0.9%. DS was associated with higher in-hospital mortality (odds ratio [OR] 1.8, 95% confidence interval [CI] 1.4 to 2.4), especially in women (OR 2.4, 95% CI 1.7 to 3.4). DS/CHD admissions were more commonly associated with hypothyroidism (OR 7.7, 95% CI 6.6 to 9.0), dementia (OR 82.0, 95% CI 32 to 213), heart failure (OR 2.2, 95% CI 1.9 to 2.5), pulmonary hypertension (OR 2.5, 95% CI 2.2 to 2.9), and cyanosis or secondary polycythemia (OR 4.6, 95% CI 3.8 to 5.6). Conversely, DS/CHD hospitalizations were less likely to include cardiac procedures or surgery (OR 0.3, 95% CI 0.2 to 0.4) and were associated with lower charges (1,177 vs 1,371, p <0.0001) compared to non-DS/CHD admissions. In conclusion, DS/CHD hospitalizations represent a decreasing proportion of admissions for adults with CHD typical of DS; patients with DS/CHD are more likely to die during hospitalization but less likely to undergo a cardiac procedure.
Life expectancy for adults with Down syndrome was calculated from data for 1610 liveborn affected individuals identified in over 1,500,000 consecutive live births in British Columbia from 1908 to 1981. Survival to 68 years of age, predicted from the available data, is better than in previous estimates, but is still much poorer than for the general population: about 44.4% and 13.6% of liveborn Down syndrome individuals will survive to 60 and 68 years, respectively, compared with 86.4% and 78.4% of the general population.
The objective of this study was to develop a prospectively applicable method for classifying comorbid conditions which might alter the risk of mortality for use in longitudinal studies. A weighted index that takes into account the number and the seriousness of comorbid disease was developed in a cohort of 559 medical patients. The 1-yr mortality rates for the different scores were: "0", 12% (181); "1-2", 26% (225); "3-4", 52% (71); and "greater than or equal to 5", 85% (82). The index was tested for its ability to predict risk of death from comorbid disease in the second cohort of 685 patients during a 10-yr follow-up. The percent of patients who died of comorbid disease for the different scores were: "0", 8% (588); "1", 25% (54); "2", 48% (25); "greater than or equal to 3", 59% (18). With each increased level of the comorbidity index, there were stepwise increases in the cumulative mortality attributable to comorbid disease (log rank chi 2 = 165; p less than 0.0001). In this longer follow-up, age was also a predictor of mortality (p less than 0.001). The new index performed similarly to a previous system devised by Kaplan and Feinstein. The method of classifying comorbidity provides a simple, readily applicable and valid method of estimating risk of death from comorbid disease for use in longitudinal studies. Further work in larger populations is still required to refine the approach because the number of patients with any given condition in this study was relatively small.
Patterns and correlates of service utilization by adults with a developmental disability were examined using data from 831 mothers of an adult child with a developmental disability. A modified Andersen model of health services was used to examine service utilization in seven domains. Multinomial logistic regression revealed that predictors of services received as well as predictors of unmet need for services varied by service. Findings emphasize the importance of considering predisposing characteristics, enabling resources, and need as well as service in order to understand patterns of service utilization.
Based on standard social benefit registers, the prevalence of intellectual disability (ID) in Finland is estimated to be 0.6%, while epidemiological surveys yield 1.1%. Combining several registers, our aim was to find a more reliable estimate of the prevalence of ID, especially among children and adolescents. This is important when special or inclusive general services are planned to meet the various needs of people with ID.
A survey based on eight national health and social benefit registers.
Combining different registers yielded a mean ID prevalence of 0.70% (95% CI 0.69-0.70%), with marked differences according to sex and age group (range 0.38-0.96%). Capture-recapture analysis gave higher prevalence estimates (range 0.57-1.08%).
When several health and social benefit registers are surveyed, the estimated prevalence of ID increases, approaching that obtained in epidemiological surveys.