Housing and Long-Term Services and Supports for People With Intellectual or Developmental Disabilities From Racially and Culturally Minoritized Communities.

This article describes research on the places people with intellectual and developmental disabilities (IDD) live and disparities in housing and long-term services and supports (LTSS) outcomes for people with IDD from racially and culturally minoritized groups. It also summarizes the conclusions and recommendations of the Housing and Long-Term Services and Supports strand of the 2022 State of the Science Conference on the Intersection of Diversity, Equity and Inclusion and Supports and Services for People with IDD, identifies limitations of the available research and recommends strategies to improve research, knowledge translation, and practices.

Association Between Midpregnancy Polyunsaturated Fatty Acid Levels and Offspring Autism Spectrum Disorder in a California Population-Based Case-Control Study.

Polyunsaturated fatty acids (PUFAs) are critical for brain development and have been linked with neurodevelopmental outcomes. We conducted a population-based case-control study in California to examine the association between PUFAs measured in midpregnancy serum samples and autism spectrum disorder (ASD) in offspring. ASD cases (n = 499) were identified through the California Department of Developmental Services and matched to live-birth population controls (n = 502) on birth month, year (2010 or 2011), and sex. Logistic regression models were used to examine crude and adjusted associations. In secondary analyses, we examined ASD with and without co-occurring intellectual disability (ID; n = 67 and n = 432, respectively) and effect modification by sex and ethnicity. No clear patterns emerged, though there was a modest inverse association with the top quartile of linoleic acid level (highest quartile vs. lowest: adjusted odds ratio = 0.74, 95% confidence interval: 0.49, 1.11; P for trend = 0.10). Lower levels of total and ω-3 PUFAs were associated with ASD with ID (lowest decile of total PUFAs vs. deciles 4-7: adjusted odds ratio = 2.78, 95% confidence interval: 1.13, 6.82) but not ASD without ID. We did not observe evidence of effect modification by the factors examined. These findings do not suggest a strong association between midpregnancy PUFA levels and ASD. In further work, researchers should consider associations with ASD with ID and in other time windows.

Mentally Fit: Negotiating the Boundaries of Cognitive Disability.

Why do some people's minds seem conspicuous, disabled, and ill-fitting in some contexts and not others? This special issue presents articles about people in Jordan, Uganda, the United Kingdom and the United States who live with Down syndrome, autism, intellectual disabilities, cerebral palsy, or histories of brain injuries. We focus on the disjunctive encounters between these individuals' minds and the varied relational processes in their surrounding social world in order to understand why different mental characteristics become points of concern and comparison at some points and not others - and thus to raise questions about how "fitting in" works altogether.

What Escapes Persuasion: Why Intellectual Disability Troubles 'Dependence' in Liberal Societies.

What expectations about the mind do people with intellectual disabilities depart from? A dominant argument maintains that their mental dependence troubles liberal relations premised upon a myth of autonomy. By analyzing the centrality of persuasion in a home for adults with intellectual disabilities in the UK, I ask instead about the psychological assumptions made by relationships of care. Persuasion aims to cultivate, not their independence from care but rather, a recognition of their dependence upon it. Persuasive care's repeated failure suggests an alternative answer to the question: people with intellectual disabilities are too independent-minded for this form of dependence.

Koolen-de Vries syndrome in the first adulthood patient of Southern India ancestry.

Koolen-de Vries syndrome (KdVS, MIM#610443) is a rare malformation condition mainly characterized by cognitive impairment in association with craniofacial and visceral anomalies. The core phenotype is caused by mutations in the chromatin remodeler KANSL1 (MSL1V1, KIAA1267, KAT8 Regulatory NSL Complex Subunit 1, MIM#612452), which maps to 17q21.31 critical genomic region (Koolen et al., Nature Genetics 2012;44:639-641). Considering its molecular basis, KdVS is included in the group of Developmental Disorders of Chromatin Remodeling (DDCRs), also termed chromatinopathies. We describe the first KdVS patient of Southern India ethnicity, harboring the typical de novo 17q21.31 microdeletion, including KANSL1. Observed facial features and congenital anomalies are in line with the already reported KdVS phenotype, suggesting that phenotypic features are consistent across different ethnicities.

Comparing Motor Competence of Sex- and Age-Matched Youth With Intellectual Disability From Brazil and the United States.

Youth with intellectual disabilities (IDs) demonstrate below-criteria motor competence (MC) compared with typically developing (TD) youth. Whether differences in MC exist for youth with ID from different countries is unknown. This study examined the MC of youth with ID from Brazil (BR) and the United States (US) and compared it with norms for TD youth as established by the Bruininks-Oseretsky Test of Motor Proficiency (BOT-2). The authors measured 19 BOT-2 test items for bilateral coordination, balance, and upper limb coordination of 502 youth (BR = 252, US = 250) with ID (6-21 years). Raw scores were converted to %ceiling (percentile of highest expected scores). For all test items, no significant differences were seen between BR and US participants in %ceiling scores. Participants from both countries demonstrated equivalent to slightly below BOT-2 norms in 14 of the 19 test items, with lowest scores seen in contralateral synchronizing bilateral coordination, balancing on one leg, and ball handling.

Psychosocial and cultural needs of children with intellectual disability and their families among the Syrian refugee population in Turkey.

BACKGROUND: Turkey is the country hosting the largest number of refugees from Syria, with currently 3 571 175 million persons. The general health needs of the refugees are being addressed; however, people with intellectual disabilities (IDs), particularly children, are relatively missed. The aim of this study was to identify medical, psychological and social needs of children with ID and their families, among the Syrian refugee population in Turkey, and to define psychosocial and cultural needs for planning of future services. METHODS: One hundred forty-two children (67.6% men; mean age 90.5 months) diagnosed with intellectual disorders were included in the study. Family Needs Survey, with additional open-ended and close-ended questions, was used to evaluate family needs. Items from Developmental Disabilities Profile-2 were used to evaluate and screen cognitive, motor and language development as well as medical concerns and behavioural problems. Data on sociodemographic characteristics were also collected. RESULTS: The highest needs were identified in information and financial needs domains. Other indicated needs were on child care and community services domains. The least indicated items were on family and social support and explaining to others domains. An average of 63.5% of the respondents definitely agreed with the Family Needs Survey items. The overall level of identification of need items was higher than that in some previous studies, indicating the level of unmet needs of the studied population. Family income, parents' employment and parents' education were not significantly associated with unmet family needs. Special education services were unreachable for most of the families in the study. CONCLUSIONS: Families reported the highest needs in information and financial needs domains. The overall level of identification of needs was higher than that in some previous studies, indicating the level of unmet needs of the studied population. The majority of the parents reported that they had sufficient family and social support, which might be a protective factor for parental mental health. Parents' Turkish fluency was very significantly associated with every domain of unmet needs. Language barriers and translation problems had significant negative effects on families, as confirmed by the answers to open-ended questions. Another important factor identified was access to service professionals. Special education services, sorely needed for most of the families, could not always be reached. Although the progress of children who had received special education was not very encouraging, it was better than those who did not receive it. The first implication of the study is that increasing Turkish proficiency or providing high-quality and consistent translation services is vital for this category of children with ID. The second implication is that information and financial needs must be met with priority. Information must be tailored for each child's needs and developmental level. Special education and physical therapy must be more accessible and at higher quality.

'We need to share our stories': the lives of Pakistanis with intellectual disability and their guardians.

INTRODUCTION: The experiences of Pakistanis with intellectual disabilities (IDs) and their family members have been underexplored empirically. METHOD: The present study sought to address this gap by understanding the lives of five Special Olympics Pakistan athletes and their guardians through PhotoVoice. FINDINGS: Through thematic analysis, we present the primary theme concerning Pakistan's cultural context that provides an empirical exploration of cultural beliefs about intellectual disability, cultural expectations and support received by people with intellectual disabilities and their guardians. DISCUSSION: We discuss implications for research and practice.

Exploring health and wellness among Native American adults with intellectual and/or developmental disabilities and their family caregivers.

BACKGROUND: Adults with intellectual and/or developmental disabilities (IDD) experience health inequities, and those who also identify as a member of an ethnic minority group face additional health inequities. In the United States, a majority of adults with IDD continue to be supported by family caregivers making their health equity also important. The purpose of this study was to explore how Native American adults with IDD and their family caregivers experience health and wellness. METHOD: This community-engaged research was guided by a Community Advisory Board (CAB) with study participants completing a Photovoice project. RESULTS: Participants identified individual, family and community level influences on health and wellness including the importance of participation in meaningful activities and connection to culture. CONCLUSIONS: In order to address health inequities, more research is needed to understand health and wellness from the unique perspectives of individuals with IDD and those from racial and ethnic minority groups.

Racial and Ethnic Disparities in Autism-Related Health and Educational Services.

OBJECTIVE: The objective of this study is to measure racial and ethnic disparities in autism-related services among U.S. children with parent-reported autism spectrum disorder (ASD). METHODS: Using the 2011 Survey of Pathways to Diagnosis and Services, we analyzed parent-reported data on 1420 children with current ASD in the nationally representative sample. An estimation method consistent with the Institute of Medicine's definition of health care disparities is used to measure racial and ethnic disparities. RESULTS: The findings revealed Latino-white disparities in the percentage of children currently using school-based occupational and physical therapy and Latino-white and "other race"-white disparities in the percentage of children using physical therapy outside of school. There were no statistically significant black-white disparities. Instead, the study found that the percentage of black children with ASD receiving school-based services was 8 points higher than that of white children (p < 0.04). Factors unrelated to the need for autism services were largely unassociated with the receipt of services. CONCLUSION: The findings provide a partial baseline and identify a need for further examination of the source of existing disparities and the lack of disparities found for specific services and minority groups.

Factors Influencing Low Prevalence of Neurodevelopmental Disabilities Among US Hispanic/Latino Children.

OBJECTIVES: Hispanic/Latino (H/L) children have lower prevalence of neurodevelopmental disabilities (NDD) than other groups. The explanations for this are complex, but may be related to nativity, language barriers, and lack of access to and utilization of healthcare. Previous research focused on how these factors affect children with NDD, but little research has jointly examined whether these factors predict NDD. This study examines whether social and environmental factors explain low prevalence of NDD in this population. METHODS: This study uses nationally representative Integrated Public Use Microdata Series National Health Interview Survey data (N = 200,622) and multivariate logistic regression analysis to compare NDD prevalence in white and H/L children (average age of 10.2), and examines whether nativity, healthcare access, healthcare utilization, and language barriers explain this disability disparity. RESULTS: Findings reveal that the H/L NDD disparity is not explained by differences in access to or utilization of healthcare, or as a result of language differences that may create barriers to NDD diagnosis. While H/L children whose sampled adult was born in the USA have lower rates of NDD than whites, H/Ls whose sampled adult were not born in the USA have even lower probability of NDD than H/Ls who were born in the USA. CONCLUSIONS: These findings may be a result of cultural differences in knowledge or understanding of what constitutes a disability or the result of differential treatment within the healthcare system among H/Ls. The findings underscore the importance of accessible and culturally appropriate health and clinical care interventions among H/L communities.

Service use and perceptions of service effectiveness by parents of individuals with intellectual disabilities: comparing Jewish and Arab Israeli parental caregivers.

BACKGROUND: The relationship between ethnicity, service use and perceptions of service effectiveness is inconclusive. This study examined differences in service use and perceptions of service effectiveness between Israeli Jewish (Jewish) and Israeli Arab (Arab) parental caregivers of individuals with intellectual disabilities and dual diagnosis of psychopathology. METHODS: Parental caregivers (n = 186) of individuals with intellectual disabilities or dual diagnosis, aged 10 to 30 years, completed a self-report questionnaire. RESULTS: Arab parental caregivers perceived health services to be more accessible than did Jewish caregivers, but there was no difference between the two groups in the use of the services. Overall, greater enabling factors and accessibility were associated with higher use of education and social services. No differences were found between the groups in their perceptions of service effectiveness. CONCLUSION: Arab family caregivers use education and social services less than do their Jewish peers, possibly because they have fewer enabling resources. The finding that both groups reported similar use of health services may be explained by a shared perception that informal help may not be suitable for dealing with situations of psychopathology. The similar perceptions of service effectiveness may be explained by extensive services available in Israel, to the satisfaction of both groups, or by the fact that participants perceived these services as their only alternative, and therefore fear losing them.

Mental health among Sami people with intellectual disabilities.

The first living condition-survey among people with intellectual disability in Sami areas in Norway was conducted in 2017. The purpose of this article is to present and discuss results from the living-condition study, with a focus on the results related to mental health and bullying as a risk factor for poor mental health among people with intellectual disability and a Sami background. We have conducted a questionnaire survey among people with intellectual disability in Sami areas, with and without a Sami background (N = 93). People with intellectual disability have poorer mental health compared to the population in general and those with Sami background have the poorest mental health. Bullying is one of several factors that increase the risk of poor mental health among people with intellectual disability and Sami background. Having a Sami background makes people with intellectual disability more disposed to poor mental health.

Ghanaian traditional and faith healers' explanatory models of intellectual disability.

BACKGROUND: The use of traditional/alternative medicine for health care in Africa has been examined for various conditions. However, there is limited research about traditional/alternative health care for intellectual disability. The present authors explored the explanatory models (EMs) of intellectual disability held by traditional/faith healers in Ghana. METHODS: Using a case vignette, the present authors conducted semi-structured interviews with 36 traditional/faith healers in Accra, Ghana. Using the EMs of illness framework as a guide, participants were asked questions to examine their beliefs about the nature, causes, course and recommended treatment of intellectual disability. RESULTS: The healers' causal explanations included maternal negligence during pregnancy and spiritual factors. They also believed that intellectual disability was a congenital, lifelong condition which could not be cured. CONCLUSIONS: Unlike other conditions which traditional healers claimed to cure, participants admitted that intellectual disability was not curable. The present authors suggest that the findings highlight opportunities that exist for collaboration between biomedicine and traditional medicine.

A qualitative study about quality of life in Brazilian families with children who have severe or profound intellectual disability.

BACKGROUND: This study investigated the psychocultural perspectives concerning family quality of life among Brazilian families with children who have severe or profound intellectual disability. METHODS: Individual in-depth semi-structured interviews conducted with 15 mothers, selected by convenience, were analysed using a categorical thematic analysis technique. The themes were examined to allow for an interpretative approach of the results. RESULTS: Mothers revealed that their children with disabilities had insufficient access to services and support related to health care, transportation and recreation. Family quality of life was negatively affected by financial restrictions and social interaction difficulties. Caring for a child with disabilities seemed to be centred on the mother and religious coping appeared as a common psychological adjustment strategy. CONCLUSIONS: Improving emotional and psychological cares, as well as social and practical measures comprising income support and access to appropriate health care, were inferred to be the mothers' priorities to improve their families' quality of life.

Racial and ethnic disparities in birth outcomes and labour and delivery-related charges among women with intellectual and developmental disabilities.

BACKGROUND: Women with intellectual and developmental disabilities (IDD) in the USA are bearing children at increasing rates. However, very little is known whether racial and ethnic disparities in birth outcomes and labour and delivery-related charges exist in this population. This study investigated racial and ethnic disparities in birth outcomes and labour and delivery-related charges among women with IDD. METHODS: The study employed secondary analysis of the 2004-2011 Healthcare Cost and Utilization Project National Inpatient Sample, the largest all-payer, publicly available US inpatient healthcare database. Hierarchical mixed-effect logistic and linear regression models were used to compare the study outcomes. RESULTS: We identified 2110 delivery-associated hospitalisations among women with IDD including 1275 among non-Hispanic White women, 527 among non-Hispanic Black women and 308 among Hispanic women. We found significant disparities in stillbirth among non-Hispanic Black and Hispanic women with IDD compared with their non-Hispanic White peers [odds ratio = 2.50, 95% confidence interval (CI): 1.16-5.28, P < 0.01 and odds ratio = 2.53, 95% CI: 1.08-5.92, P < 0.01, respectively]. There were no racial and ethnic disparities in caesarean delivery, preterm birth and small-for-gestational-age neonates among women with IDD. The average labour and delivery-related charges for non-Hispanic Black and Hispanic Women with IDD ($18 889 and $22 481, respectively) exceeded those for non-Hispanic White women with IDD ($14 886) by $4003 and $7595 or by 27% and 51%, respectively. The significant racial and ethnic differences in charges persisted even after controlling for a range of individual-level and institutional-level characteristics and were 6% (ln(ß) = 0.06, 95% CI: 0.01-0.11, P < 0.05) and 9% (ln(ß) = 0.09, 95% CI: 0.03-0.14, P < 0.01) higher for non-Hispanic Black and Hispanic Women with IDD compared with non-Hispanic White women with IDD. CONCLUSIONS: Our findings highlight the need for an integrated approach to the delivery of comprehensive perinatal services for racial and ethnic minority women with IDD to reduce their risk of having a stillbirth. Additionally, further research is needed to examine the causes of racial and ethnic disparities in hospital charges for labour and delivery admission among women with IDD and ascertain whether price discrimination exists based on patients' racial or ethnic identities.

A novel SLC6A8 mutation associated with intellectual disabilities in a Chinese family exhibiting creatine transporter deficiency: case report.

BACKGROUND: X-linked creatine transporter deficiency (OMIM#300036,CRTR-D) is characterized by cerebral creatine deficiency, intellectual disabilities, severe speech impairment, seizures and behavioral problems. Mutations in the creatine transporter gene SLC6A8, a member of the solute-carrier family 6 mapped to Xq28, have been reported to cause the creatine transporter deficiency. CASE PRESENTATION: The proband presented at 5 yrs. 1 month of age with delays in intellectual and development, seizures and behavioral problems. A novel missense mutation, c.1181C > A (p.Thr394Lys), in the SLC6A8 gene (NM_005629.3) was detected via targeted exome sequencing, and then validated by Sanger sequencing. Multiple in silico variant effect analysis methods, including SIFT, PolyPhen2, PROVEAN, and Mutation Taster predicted that this variant was likely damaging or diseasing-causing. This hemizygous variation was also identified in the affected brother with the same clinical condition and inherited from the heterozygous carrier mother. The diagnosis was suggested by increased urinary creatine/creatinine (Cr:Crn) ratio and markedly reduced creatine content peak by brain proton magnetic resonance spectroscopy (MRS). The proband's mother became pregnant with a 3rd sibling, in whom the Sanger sequencing result of c.1181C > A was negative. CONCLUSION: The novel mutation c.1181C > A in the SLC6A8 gene reported in a Chinese family has expanded the mutation spectrum of CRTR-D. The combination of powerful new technologies such as targeted exome sequencing with thorough systematic clinical evaluation of patients will improve the diagnostic yield, and assist in genetic counselling and prenatal diagnosis for suspected genetic disorders.

Novel MEF2C point mutations in Chinese patients with Rett (-like) syndrome or non-syndromic intellectual disability: insights into genotype-phenotype correlation.

BACKGROUND: MEF2C (Myocyte-specific enhancer factor 2C) has been associated with neurodevelopmental disorders. This study aimed at delineating the clinical profiles of MEF2C gene mutations. METHODS: In total, 112 Chinese patients with intellectual disability (ID) were recruited, including 44 patients presented with Rett syndrome (RTT) or RTT-like syndrome, and 68 patients with non-syndromic ID. Targeted next-generation sequencing (NGS) was performed. Detailed clinical information was collected. RESULTS: Five heterozygous MEF2C gene mutations were identified, of which three were novel. The MEF2C mutant rate was 4.5% (5/112) in total, and 6.8% (3/44) in the RTT (-like) cohort. All patients with MEF2C gene mutation presented with cognitive impairment, gross motor delay, speech disorder and autistic features. Four patients had epilepsy, which responded well to antiepileptic drugs. One female was diagnosed with classical RTT, two females with RTT-like syndrome, and two males with non-syndromic ID. Generally, the phenotype of two males with relatively downstream mutations (c.565C > T, p.Arg 189*; c.766C > T, p.Arg 256*) was milder than that of three females with upstream mutations (c.48C > G, p.Asn16Lys; c.334G > T, p.Glu112* and c.403-1G > T). CONCLUSIONS: Our findings expanded the current understanding of the consequences of MEF2C dysfunctions, especially MEF2C point mutations. MEF2C mutations are associated with a broad clinical spectrum, ranged from classical RTT to non-syndromic ID. Through our study, it can be inferred that there is correlation between the phenotype and MEF2C-genotype, the mutation site. Overall, the MEF2C gene mutational analysis should be performed in ID cohort, especially in patients with features overlapped with RTT.

"We Had to Keep Pushing": Caregivers' Perspectives on Autism Screening and Referral Practices of Black Children in Primary Care.

Black children with autism spectrum disorder (ASD) are diagnosed later than their White peers, are more likely to be misdiagnosed, and are less likely to receive early intervention services or a developmental evaluation by three years old. Using a grounded theory approach, we solicited the perspectives of parents and other primary caregivers of Black children with ASD on barriers and facilitators to ASD screening and referrals in primary care. A socioeconomically diverse sample of 22 female caregivers participated. Four themes emerged. First, while some caregivers noted their child's primary healthcare providers facilitated a timely ASD diagnosis, other participants reported these providers ignored early concerns about child developmental delays. Second, many participants felt racial bias negatively impacted caregiver-primary healthcare provider interactions. Third, legal/custodial issues slowed caregivers' abilities to follow up on referrals from their primary healthcare providers. Finally, caregivers described denial, shame, and stigma relating to ASD in the Black community as possible factors for delayed follow up to referrals. Differences based on socioeconomic status are discussed. Efforts to improve family-centered, culturally relevant care for all Black caregivers raising children with or at-risk for ASD are needed, particularly for those families experiencing the multiple effects of poverty.

Family and community in the lives of UK Bangladeshi parents with intellectual disabilities.

BACKGROUND: Little is known about the lives of parents with intellectual disabilities from minority ethnic communities. Previous research suggests that what it means to live with intellectual disabilities varies across cultural contexts. The current research aimed to explore how cultural values and practices impact upon the experiences of parents with intellectual disabilities within the Bangladeshi community in London, England. METHOD: Six members of the Bangladeshi community, four Bangladeshi parents with intellectual disabilities and four of their family members were interviewed. Thematic analysis was used to identify key themes. RESULTS: Both parenting and intellectual disability are thought about in this community in ways that make parenting more accessible for people with learning disabilities, but also create tensions to be negotiated. CONCLUSIONS: Bangladeshi family carers face dilemmas balancing the benefits and risks of promoting parenting for sons and daughters with intellectual disabilities, particularly in the context of service principles of autonomy and informed consent.