Disability, Hospital Care, and Cost: Utilization of Emergency and Inpatient Care by a Cohort of Children with Intellectual and Developmental Disabilities.

OBJECTIVE: To use medical claims data to determine patterns of healthcare utilization in children with intellectual and developmental disabilities, including frequency of service utilization, conditions that require hospital care, and costs. STUDY DESIGN: Medicaid administrative claims from 4 states (Iowa, Massachusetts, New York, and South Carolina) from years 2008-2013 were analyzed, including 108 789 children (75 417 male; 33 372 female) under age 18 years with intellectual and developmental disabilities. Diagnoses included cerebral palsy, autism, fetal alcohol syndrome, Down syndrome/trisomy/autosomal deletions, other genetic conditions, and intellectual disability. Utilization of emergency department (ED) and inpatient hospital services were analyzed for 2012. RESULTS: Children with intellectual and developmental disabilities used both inpatient and ED care at 1.8 times that of the general population. Epilepsy/convulsions was the most frequent reason for hospitalization at 20 times the relative risk of the general population. Other frequent diagnoses requiring hospitalization were mood disorders, pneumonia, paralysis, and asthma. Annual per capita expenses for hospitalization and ED care were 100% higher for children with intellectual and developmental disabilities, compared with the general population ($153 348 562 and $76 654 361, respectively). CONCLUSIONS: Children with intellectual and developmental disabilities utilize significantly more ED and inpatient care than other children, which results in higher annual costs. Recognizing chronic conditions that increase risk for hospital care can provide guidance for developing outpatient care strategies that anticipate common clinical problems in intellectual and developmental disabilities and ensure responsive management before hospital care is needed.

Adaptive innovations to provide services to children with developmental disabilities during the COVID-19 pandemic.

Children with developmental disabilities are experiencing significant challenges to service access due to suspension of in-person assessments during the current COVID-19 pandemic. Telehealth is rapidly becoming the new service delivery model, which presents a unique opportunity for innovation in care that could be beneficial in the post-pandemic period. For example, using a combination of in-home video and telehealth options could form the first step in developmental assessment, allowing children to receive the necessary supports without delay. Recent telehealth funding is welcome but additional Medicare items for joint consultations including general practitioners (GPs), and paediatric, mental health and allied health professionals is critical.

Health Care Use and Spending of Pediatric Patients With an Intellectual or Developmental Disability.

BACKGROUND: Health care costs and utilization for those with an intellectual or developmental disability (IDD) have been shown to be higher than the general population. OBJECTIVE: To investigate the services that contribute to higher costs and utilization among noninstitutionalized children with an IDD. DESIGN: Matched case-control secondary analysis of the 2000-2017 Medical Expenditure Panel Survey. Pediatric (age 0-21) patients with an IDD were matched to non-IDD subjects. Health care utilization and costs were evaluated with zero-inflated negative binomial regressions and generalized linear models, respectively. MEASURES: Outcome measures included high-acuity health care utilization [ie, emergency department (ED) visits and hospital admissions], and cost outcomes for total spending, ED use, hospitalization, medications, office visits, home health, and physical therapy. RESULTS: There was no statistical difference in utilization of EDs among the 2 groups though subjects with an IDD showed more hospitalizations than their matched cohort (incidence rate ratios=1.63, P=0.00). Total health care spending was higher among patients with an IDD (coefficient=$5831, P=0.00). Pediatric spending was higher in all measures except for ED. The biggest discrepancies in spending were seen in home health (coefficient=$2558, P=0.00) and outpatient visits (coefficient=$1180, P=0.00). CONCLUSIONS: Pediatric patients with an IDD had higher health care spending and utilization than non-IDD subjects in all categories except for ED use.

High health care costs among adults with intellectual and developmental disabilities: a population-based study.

OBJECTIVE: While it is generally accepted that adults with intellectual and developmental disabilities (IDDs) use health services to a greater extent than the general population, there is remarkably little research that focuses on the costs associated with their health care. Using population-based data from adults with IDD in Ontario, this study aimed to estimate overall health care costs, classify individuals into high and non-high cost categories and describe differences in the demographics, clinical profiles and health care use patterns between these groups. DESIGN: A retrospective cohort study based in Ontario, Canada, was conducted with the use of linked administrative health data. METHODS: A costing algorithm developed for the general population in Ontario was applied to estimate health care costs of adults with IDD under age 65 for 2009 and 2010. Individuals were categorised into two groups according to whether their total annual health care costs were among the highest decile in the general population. These groups were compared on demographic and clinical variables, and relative mean costs for six types of health care services in the two groups were computed. In addition, we computed the proportion of individuals who remained in the high cost group over 2 years. RESULTS: Among adults with IDD, 36% had annual health care expenditures greater than $2610 CAD (top decile of all Ontario adults under 65). These individuals were more likely to be female, to be in the oldest age groups, to live in group homes and to be receiving disability income support than individuals whose expenditures were below the high cost threshold. In addition, they had higher rates of all the physical and mental health conditions studied. Greatest health care expenses were due to hospitalisations, especially psychiatric hospitalisations, continuing care/rehabilitation costs and medication costs. The majority of individuals whose health care costs placed them in the high cost category in 2009 remained in that category a year later. DISCUSSION: Adults with IDD are nearly 4 times as likely to incur high annual health care costs than those without IDD. Individuals with IDD and high health care costs have unique health and demographic profiles compared with adults with IDD whose annual health care costs are below the high cost threshold. Attending to their health care needs earlier in their health care trajectory may be an opportunity to improve health and reduce overall health care costs. It is important that we explore how to best meet their needs. Models proposed to meet the needs of adults with high health care costs in the general population may not apply to this unique group.

The relationship of undernutrition/psychosocial factors and developmental outcomes of children in extreme poverty in Ethiopia.

BACKGROUND: Extreme poverty is severe deprivation of basic needs and services. Children living in extreme poverty may lack adequate parental care and face increased developmental and health risks. However, there is a paucity of literature on the combined influences of undernutrition and psychosocial factors (such as limited play materials, playground, playtime, interactions of children with their peers and mother-child interaction) on children's developmental outcomes. The main objective of this study was, therefore, to ascertain the association of developmental outcomes and psychosocial factors after controlling nutritional indices. METHODS: A community-based cross-sectional study design was used to compare the developmental outcomes of extremely poor children (N = 819: 420 girls and 399 boys) younger than 5 years versus age-matched reference children (N = 819: 414 girls and 405 boys) in South-West Ethiopia. Using Denver II-Jimma, development in personal-social, language, fine and gross motor skills were assessed, and social-emotional skills were evaluated using the Ages and Stages Questionnaires: Social-Emotional (ASQ: SE). Nutritional status was derived from the anthropometric method. Independent samples t-test was used to detect mean differences in developmental outcomes between extremely poor and reference children. Multiple linear regression analysis was employed to identify nutritional and psychosocial factors associated with the developmental scores of children in extreme poverty. RESULTS: Children in extreme poverty performed worse in all the developmental domains than the reference children. Among the 819 extremely poor children, 325 (39.7%) were stunted, 135 (16.5%) were underweight and 27 (3.3%) were wasted. The results also disclosed that stunting and underweightness were negatively associated with all the developmental skills. After taking into account the effects of stunting and being underweight on the developmental scores, it was observed that limited play activities, limited child-to-child interactions and mother-child relationships were negatively related mainly to gross motor and language performances of children in extreme poverty. CONCLUSION: Undernutrition and psychosocial factors were negatively related to the developmental outcomes, independently, of children living in extreme poverty. Intervention, for these children, should integrate home-based play-assisted developmental stimulation and nutritional rehabilitation.

Therapy Use for Children With Developmental Conditions: Analysis of Colorado Medicaid Data.

STUDY PURPOSE: To examine therapy use and spending for Medicaid-enrolled infants and toddlers with developmental conditions. METHODS: Sample infants and toddlers had a diagnosis (eg, cerebral palsy) or developmental delay (DD). Colorado Children's Medicaid administrative outpatient therapy claims (2006-2008) were used to estimate differences, by condition type and number of comorbid chronic conditions (CCCs), of any physical therapy (PT)/occupational therapy (OT) and Medicaid PT/OT spending. RESULTS: The sample included 20 959 children. Children with at least 2 CCCs had higher odds of PT/OT than children with no CCC. Children with DD had 12-fold higher odds of having any PT/OT compared with children with diagnosis. Children with a DD and 2 CCCs had the highest PT/OT spending. CONCLUSIONS: Medicaid PT/OT use and spending are higher for children with more CCCs and those with DD because children with DD receive more specialized PT/OT.

Developmental Disabilities Program. Final rule.

This rule implements the Developmental Disabilities Assistance and Bill of Rights Act of 2000. The previous regulations were completed in 1997 before the current law was passed. The rule will align the regulations and current statute and will provide guidance to AIDD grantees.

The metabolic evaluation of the child with an intellectual developmental disorder: diagnostic algorithm for identification of treatable causes and new digital resource.

Intellectual developmental disorders (IDD), characterized by significant impairment of cognitive functions, with limitations of learning, adaptive behavior and skills, are frequent (2.5% of the population affected) and present with significant co-morbidity. The burden of IDD, in terms of emotional suffering and associated health care costs, is significant; prevention and treatment therefore are important. A systematic literature review, updated in 2013, identified 89 inborn errors of metabolism (IEMs), which present with IDD as prominent feature and are amenable to causal therapy. Therapeutic effects include improvement and/or stabilization of psychomotor/cognitive development, behavior/psychiatric disturbances, seizures, neurologic and systemic manifestations. The levels of available evidence for the various treatments range from Level 1b, c (n=5); Level 2a, b, c (n=14); Level 4 (n=53), and Levels 4-5 (n=27). For a target audience comprising clinical and biochemical geneticists, child neurologists and developmental pediatricians, five experts translated....this data into a 2-tiered diagnostic algorithm: The first tier comprises metabolic "screening" tests in urine and blood, which are relatively accessible, affordable, less invasive, and have the potential to identify 60% of all treatable IEMs. The second tier investigations for the remaining disorders are ordered based on individual clinical signs and symptoms. This algorithm is supported by an App www.treatable-id.org, which comprises up-to-date information on all 89 IEMs, relevant diagnostic tests, therapies and a search function based on signs and symptoms. These recommendations support the clinician in early identification of treatable IEMs in the child with IDD, allowing for timely initiation of therapy with the potential to improve neurodevelopmental outcomes. The need for future studies to determine yield and usefulness of these recommendations, with subsequent updates and improvements to developments in the field, is outlined.

Economic impacts of environmentally attributable childhood health outcomes in the European Union.

BACKGROUND: There is increasing evidence of the role that exposure to industrial chemicals plays in the development of childhood disease. The USA and the European Union (EU) have taken divergent policy approaches to managing this issue, and economic estimates of disease costs attributable to environmental exposures in children are available in the USA but not the EU. We undertook the first economic evaluation of the impacts of childhood environmental chemical exposures in the EU. METHODS: We used a cost-of-illness approach to estimate health care system costs, and used environmentally attributable fraction modelling to estimate the proportion of childhood disease due to environmental exposures. We analysed data on exposures, disease prevalence and costs at a country level, and then aggregated costs across EU member states to estimate overall economic impacts within the EU. RESULTS: We found the combined environmentally attributable costs of lead exposure, methylmercury exposure, developmental disabilities, asthma and cancer to be $70.9 billion in 2008 (range: $58.9-$90.6 billion). These costs amounted to ~0.480% of the gross domestic product of the EU in 2008. CONCLUSIONS: Childhood chemical exposures present a significant economic burden to the EU. Our study offers an important baseline of disease costs before the implementation of Registration, Evaluation and Authorization of Chemicals, which is important for studying the impacts of this policy regime.

The economic burden of prematurity in Canada.

BACKGROUND: Preterm birth is a major risk factor for morbidity and mortality among infants worldwide, and imposes considerable burden on health, education and social services, as well as on families and caregivers. Morbidity and mortality resulting from preterm birth is highest among early (< 28 weeks gestational age) and moderate (28-32 weeks) preterm infants, relative to late preterm infants (33-36 weeks). However, substantial societal burden is associated with late prematurity due to the larger number of late preterm infants relative to early and moderate preterm infants. METHODS: The aim in this study was to characterize the burden of premature birth in Canada for early, moderate, and late premature infants, including resource utilization, direct medical costs, parental out-of-pocket costs, education costs, and mortality, using a validated and published decision model from the UK, and adapting it to a Canadian setting based on analysis of administrative, population-based data from Québec. RESULTS: Two-year survival was estimated at 56.0% for early preterm infants, 92.8% for moderate preterm infants, and 98.4% for late preterm infants. Per infant resource utilization consistently decreased with age. For moderately preterm infants, hospital days ranged from 1.6 at age two to 0.09 at age ten. Cost per infant over the first ten years of life was estimated to be $67,467 for early preterm infants, $52,796 for moderate preterm infants, and $10,010 for late preterm infants. Based on population sizes this corresponds to total national costs of $123.3 million for early preterm infants, $255.6 million for moderate preterm infants, $208.2 million for late preterm infants, and $587.1 million for all infants. CONCLUSION: Premature birth results in significant infant morbidity, mortality, healthcare utilization and costs in Canada. A comprehensive decision-model based on analysis of a Canadian population-based administrative data source suggested that the greatest national-level burden is associated with moderate preterm infants due to both a large cost per infant and population size while the highest individual-level burden is in early preterm infants and the largest total population size is in late preterm infants. Although the highest medical costs are incurred during the neonatal period, greater resource utilization and costs extend into childhood.

Incorporating life course theory and social determinants of health into the LEND curriculum.

The goal of this paper is to describe strategies for revising LEND curricula to incorporate a stronger focus on life course theory and social determinants of health (LCT/SDOH). The Maternal and Child Health Bureau (MCHB) includes a central focus on LCT/SDOH and states that a goal of Maternal and Child Health (MCH) training is to "Prepare and empower MCH leaders to promote health equity…and reduce disparities in health and health care." Two LEND programs engaged in a comprehensive process to strengthen LCT/SDOH in their curricula that included choosing content and themes and developing instructional strategies congruent with MCH Leadership Competencies and with the learning needs of LEND trainees. We describe: key elements of LCT/SDOH; the relationship of these to children with disabilities and to the MCH Leadership Competencies; LCT/SDOH resources for the LEND curriculum; a collaborative curriculum revision process for faculty; and LCT/SDOH content and themes for the LEND Curriculum and strategies for incorporating them. We present the results of our work in a format that may be used by other LEND programs undertaking curriculum revision to incorporate LCT/SDOH.

Restricting state part C eligibility policy is associated with lower early intervention utilization.

To examine if state differences in early intervention (EI) utilization can be explained by recent restrictions on EI state eligibility policy. The sample (n = 923), derived from the 2009/10 National Survey of Children with Special Health Care Needs, included CSHCN who were ages 0-3 with a developmental delay or disability that affected their function. Multi-level logistic modeling was used to describe state differences in EI utilization and to determine if narrower state eligibility policy explained these differences. EI utilization ranged from 6 to 87 % across states. Having a severe condition (ß = 0.99, SE = 0.28) and a usual source of care (ß = 0.01, SE = 0.001) was associated with higher odds of utilizing EI. Compared to a diagnosed disability, having a developmental delay (ß = -0.61, SE = 0.20) was associated with lower odds of utilizing EI. Living in a state with narrow and narrower state eligibility policy (ß = -0.18, SE = 0.06) was significantly associated with lower odds of EI utilization, and this effect was strongest for children with the most severe functional impairments. Significant state variation in EI rates exists that can be explained, in part, by the restrictiveness of state eligibility criteria. Children with the most severe functional impairments appear to be least likely to utilize EI in states with the most restrictive eligibility policies.

Setting global research priorities for developmental disabilities, including intellectual disabilities and autism.

OBJECTIVES: The prevalence of intellectual disabilities (ID) has been estimated at 10.4/1000 worldwide with higher rates among children and adolescents in lower income countries. The objective of this paper is to address research priorities for development disabilities, notably ID and autism, at the global level and to propose the more rational use of scarce funds in addressing this under-investigated area. METHODS: An expert group was identified and invited to systematically list and score research questions. They applied the priority setting methodology of the Child Health and Nutrition Research Initiative (CHNRI) to generate research questions and to evaluate them using a set of five criteria: answerability, feasibility, applicability and impact, support within the context and equity. FINDINGS: The results of this process clearly indicated that the important priorities for future research related to the need for effective and efficient approaches to early intervention, empowerment of families supporting a person with developmental disability and to address preventable causes of poor health in people with ID and autism. CONCLUSIONS: For the public health and other systems to become more effective in delivering appropriate support to persons with developmental disabilities, greater (and more targeted) investment in research is required to produce evidence of what works consistent with international human rights standards.

Resilient parenting of preschool children at developmental risk.

BACKGROUND: Given the great benefits of effective parenting to child development under normal circumstances, and the even greater benefits in the face of risk, it is important to understand why some parents manage to be effective in their interactions with their child despite facing formidable challenges. This study examined factors that promoted effective parenting in the presence of child developmental delay, high child behaviour problems, and low family income. METHOD: Data were obtained from 232 families at child age 3 and 5 years. Using an adapted ABCX model, we examined three risk domains (child developmental delay, child behaviour problems, and low family income) and three protective factors (mother's education, health, and optimism). The outcome of interest was positive parenting as coded from mother-child interactions. RESULTS: Levels of positive parenting differed across levels of risk. Education and optimism appeared to be protective factors for positive parenting at ages 3 and 5, and health appeared to be an additional protective factor at age 5. There was an interaction between risk and education at age 3; mothers with higher education engaged in more positive parenting at higher levels of risk than did mothers with less education. There was also an interaction between risk and optimism at age 3; mothers with higher optimism engaged in more positive parenting at lower levels of risk than did mothers with less optimism. The risk index did not predict change in positive parenting from age 3-5, but the protective factor of maternal health predicted positive changes. CONCLUSIONS: This study examined factors leading to positive parenting in the face of risk, a topic that has received less attention in the literature on disability. Limitations, future directions, and implications for intervention are discussed.

Childhood disabilities and the cost of developmental therapies: the serviceprovider perspective.

PURPOSE: For children with neurodevelopmental disabilities (CWNDs), early diagnosis that leads to early intervention with regular targeted therapies is critical. In Qatar, private therapy centres that address this demand often have highly exclusive prices restricting families from availing them. This paper examines the challenges faced by families with CWNDs, as well as various financial and systemic obstacles, from the vantage point of these centres, all of which culminate in an extraordinarily high disability price tag for disability families in Qatar. METHODS: This study is based on qualitative, semi-structured, and in-depth interviews with private therapy centres and developmental paediatricians. RESULTS: Therapy centre representatives expressed common struggles in lengthy and cumbersome administration and licencing procedures, difficulty in hiring and retaining high quality staff, and expenses that need to be paid to the state. From their experience, families largely struggle with delayed diagnoses that significantly slow down intervention plans and therapies as well as staggeringly high financial costs with a dearth of funding options. CONCLUSIONS: We recommend sincere engagement, dialogue, and cooperation between multiple stakeholders; a supportive ecosystem to balance and distribute the demand that includes schools and parents; as well more efficient administrative procedures and recruitment strategies.

Preterm birth and neurodevelopment: a review of outcomes and recommendations for early identification and cost-effective interventions.

This review summarizes research findings to date on neurological and health outcomes following preterm birth, tools to identify children at risk for neurodevelopmental impairment and interventions to prevent preterm birth and improve outcomes. We bring together findings from research in high- and low-income countries, with an aim to provide a global perspective on the issues. Around the world, preterm birth is rising in importance as a cause of under-five morbidity and mortality, and we project that this trend will continue over time, particularly given the lack of interventions to prevent the condition. With the development of improved screening instruments, further identification and scale up of cost-effective interventions to optimize early childhood development and accelerated research on the underlying biological mechanisms, we have an opportunity to reduce rates of neurodevelopmental impairment, particularly in countries with the highest burden.

The association between neurodevelopmental disability and economic outcomes during mid-childhood.

BACKGROUND: There is a paucity of evidence on the association between childhood disability and economic outcomes. This study estimates the economic costs and health utilities associated with neurodevelopmental impairment or serious neurodevelopmental disability during the 11th year of life. METHODS: Standardized tests of cognitive ability and clinical assessments of functional abilities were performed on participants of a whole population study of extremely preterm children and term-born controls (EPICure). Profiles of children's use of health, social and education services, and descriptions of their health status using the Health Utilities Index Mark 2 and Mark 3 health status classification systems, were extracted from detailed questionnaires completed by the main parent. Descriptive and multiple regression techniques were used to explore the association between neurodevelopmental impairment or serious neurodevelopmental disability and two sets of economic outcomes; economic costs and health utilities. RESULTS: Mean health and social service costs over the 12-month period were estimated at £577 [standard error (SE): £141] among children without neurodevelopmental impairment, £831 (£112) among children with a mild neurodevelopmental impairment, £1053 (£176) among children with a moderate neurodevelopmental impairment and £1662 (£316) among children with a severe neurodevelopmental impairment. Mean health and social service costs were estimated at £1223 (£157) and £695 (£92) among children with and without serious neurodevelopmental disability, respectively. After controlling for clinical and sociodemographic confounders, mild, moderate and severe neurodevelopment impairment increased health and social service costs by an average of £397 (95% CI: £86, £707; P= 0.012), £655 (95% CI: £167, £1144; P= 0.009) and £1065 (95% CI: -£113, £2243; P= 0.076), respectively. Cost differences were accentuated when broader public sector costs were considered. The mean (SE) Health Utilities Index Mark 3 multi-attribute utility score for the children with neurodevelopmental disability was 0.652 (0.039), compared with 0.923 (0.009) for children without neurodevelopmental disability, generating a mean difference in utility score of 0.272 (95% CI: 0.220, 0.323) that was statistically significant (P < 0.001). CONCLUSIONS: Our data should act as a significant new resource for economic analysts modelling the overall economic impact of childhood neurodevelopment impairment or disability or the cost-effectiveness of interventions targeted at neurodevelopmentally impaired or disabled children.

Africanizing the social determinants of health: embedded structural inequalities and current health outcomes in sub-Saharan Africa.

There is a growing interest in health policy in the social determinants of health. This has increased the demand for a paradigm shift within the discipline of health economics from health care economics to health economics. While the former involves what is essentially a medical model that emphasizes the maximization of individual health outcomes and considers the social organization of the health system as merely instrumental, the latter emphasizes that health and its distribution result from political, social, economic, and cultural structures. The discipline of health economics needs to refocus its energy on the social determinants of health but, in doing so, must dig deeper into the reasons for structurally embedded inequalities that give rise to inequalities in health outcomes. Especially is this the case in Africa and other low- and middle-income regions. This article seeks to provide empirical evidence from sub-Saharan Africa, including Ghana and Nigeria, on why such inequalities exist, arguing that these are in large part a product of hangovers from historically entrenched institutions. It argues that there is a need for research in health economics to embrace the social determinants of health, especially inequality, and to move away from its current mono-cultural focus.

Global perspective on early diagnosis and intervention for children with developmental delays and disabilities.

Low- and middle-income countries are experiencing a significant reduction in mortality of children under 5 years of age. This reduction is bringing in its wake large numbers of surviving children with developmental delays and disabilities. Very little attention has been paid to these children, most of whom receive minimal or no support. Thus, there is an urgent need to recognize that improving the quality of life of the survivors must complement mortality reduction in healthcare practice and programs. The incorporation of early evaluation and intervention programs into routine pediatric care is likely to have the most impact on the quality of life of these children. We therefore call for leadership from practitioners, governments, and international organizations to prioritize regular childhood developmental surveillance for possible delays and disabilities, and to pursue early referral for intervention.

Individual and system-related factors associated with the costs of intellectual and developmental disabilities.

The factors associated with the cost of intellectual and developmental disabilities are the prevalence of pervasive developmental disorders, limited access to specialized services, the high cost of prescription medication, strained relationships with providers, and not enough latitude when it comes to selection of services. These factors work together to create an environment that is either conducive to or prohibitive of quality patient outcomes and improved perceptions of publicly-provided health care. Improving any of these factors would undoubtedly lower the overall annual cost. However, more research is required to understand how these factors affect both cost and perceptions. The factors addressed in this paper are the difference between mental healthcare and general healthcare; the prevalence of intellectual and developmental disabilities; the sociological impact of Medicaid services; out-of-pocket expenses; use of specialized mental health services; access to specialized services; and patients' relationship with providers.