Definition, assessment and management of frailty for people with intellectual disabilities: A scoping review.

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.

Understanding COVID-19 infection among people with intellectual and developmental disabilities using machine learning.

BACKGROUND: People with intellectual and developmental disabilities (IDD) were disproportionately affected by the COVID-19 pandemic. Predicting COVID-19 infection has been difficult. OBJECTIVE: We sought to address two research questions in this study: 1) to assess the overall utility of a machine learning model to predict COVID-19 diagnosis for people with IDD, and 2) to determine the primary predictors of COVID-19 diagnosis in a random sample of Home and Community Based Services users in one state. METHODS: We merged three major IDD-specific datasets (National Core Indicators, Supports Intensity Scale, Medicaid HCBS expenditures) from one state to create one combined dataset for analyses that included more than 700 variables. We then built a random forest machine learning algorithm to predict COVID-19 diagnosis and to explore the top predictors of such a diagnosis, when present. RESULTS: Our algorithm predicted COVID-19 diagnosis in a random sample of HCBS users with IDD with 62.5% accuracy. The top predictors of having a documented case of COVID-19 among our sample were higher age, having high overall, medical, or behavioral support needs, living in a lower-income neighborhood, total Medicaid expenditure, and higher body mass index. CONCLUSIONS: Results largely followed trends in the general population, and were largely suggestive that increased contact with other people may have exposed a person with IDD to greater COVID-19 risk.

Differences in Telemedicine Use Between People With and Without an Intellectual or Other Developmental Disability During the COVID-19 Pandemic.

Telemedicine utilization of people with an Intellectual or Other Developmental Disability (IDD) during the COVID-19 Pandemic is not well known. This study compares telemedicine utilization of those with and without IDD prior to the pandemic to after it began. Using the Utah All Payers Claims Database from 2019 to 2021, the study identified telemedicine utilization of adults aged 18 to 62 years old in 2019. Propensity score matching was used to minimize observed confounders of subjects with and without IDD in 2019. Negative binomial regression was used to identify factors that were associated with telemedicine utilization. The final number of subjects in the analysis was 18 204 (IDD: n = 6068, non-IDD: n = 12 136 based on 1:2 propensity score matching). The average (SD) age of the subjects was 31 (11.3) years old in 2019. Forty percent of the subjects were female, about 70% of subjects were covered by Medicaid in 2019. Average (SD) number of telemedicine use in 2020 (IDD: 1.96 (5.97), non-IDD: 1.18 (4.90); P < .01) and 2021 (IDD: 2.24 (6.78) vs 1.37 (5.13); P < .01) were higher for the IDD group than the non-IDD group. The regression results showed that the subjects with IDD had 56% more telemedicine encounters than those in the non-IDD group (Incidence Rate Ratio (IRR) = 1.56, P < .01). The growth of telemedicine during the COVID-19 pandemic has the potential to reduce persistent healthcare disparities in individuals with IDD. However, quality of telemedicine should be considered when it is provided to improve health of subjects with IDD.

Health equity for people with intellectual and developmental disability requires vast improvements to data collection: Lessons from the COVID-19 pandemic.

The COVID-19 pandemic drastically underscored the lack of proper health surveillance for people with intellectual and developmental disability (IDD) in the USA. This data equity failure resulted in researchers having to rely on nontraditional data sources to develop an understanding of how this population was faring during the pandemic. To begin addressing this data equity concern, in this commentary, we (1) discuss the difficulties in accessing data during the pandemic specifically related to people with IDD; (2) provide guidance regarding how existing data can be used to examine COVID-19 outcomes for people with IDD; and (3) provide recommendations for improving data collection for people with IDD in light of lessons learned during the pandemic. In sum, the data currently available to examine COVID-19 as well as other health outcomes among people with IDD are severely limited, compromising the ability to both understand and address health disparities among this population.

Quantitative assessment of tip-toe behavior in individuals with autism spectrum disorder and intellectual disability: A cross-sectional study.

The term "toe walking" describes walking on the toes with a lack of heel strike upon initiation of the stance phase of gait. In individuals with autism spectrum disorder (ASD), this phenomenon, or "tip-toe behavior" (TTB), can be present in a substantial proportion of subjects even during standing. In this study, we investigated TTB in 50 persons with ASD (age range 4-26 years). We evaluated TTB through an observational/report-based assessment protocol. Subsequently, we employed a new structured video-based coding protocol based on standardized video recordings, focusing on static and dynamic conditions. Finally, the findings of the two protocols were compared. Twenty-four subjects with TTB were identified and classified according to three functional groups: TTB1, present only during running (6 subjects); TTB2, present during walking and running (11 subjects); and TTB3, present during standing, walking, and running (7 subjects). Moreover, we found that TTB3 subjects exhibited a significantly higher quantity of TTB compared with subjects in the TTB1 and TTB2 groups during both standing and walking tests. Additionally, a high quantity of TTB in the static test was found to be related to a high quantity of TTB in the dynamic test. Variables such as age, autism severity, intellectual disability, and gender were not significantly associated with the mean percent of TTB both in static and dynamic tests in multivariate analysis. This structured video-based coding approach appears feasible and useful for assessing TTB in individuals with ASD and it has the potential to provide insights into TTB trajectories and aid in designing possible interventions.

High cardiorespiratory disease burden following a fracture among adults with intellectual disabilities.

BACKGROUND: Adults with intellectual disabilities have a greater risk for fragility fractures that begin to accumulate early in the adult lifespan, which may contribute to accelerated health declines. The objective was to determine if fragility fractures were associated with an increased 2-year rate of cardiorespiratory diseases among adults with intellectual disabilities. METHOD: This retrospective cohort study used nationwide administrative claims data from 01/01/2011-12/31/2016 from the Medicare fee-for-service database. 2-year incidence of cardiorespiratory diseases were compared between adults ≥18 years old with intellectual disabilities with (n = 6183) vs. without (n = 67,842) an incident fragility fracture after confounder adjustment using Cox regression. RESULTS: Fracture at the vertebral column, hip, non-proximal femur, tibia/fibula, and multiple sites had an elevated hazard ratio (HR) compared to those with no fracture for pneumonia, respiratory failure, heart failure, and cerebrovascular disease (HR range, 1.15-2.09, all P < 0.05), while humerus and radius/ulna fracture were associated with an elevated HR for congestive heart failure and cerebrovascular disease (HR range, 1.38-1.72, all P < 0.05). CONCLUSIONS: Fragility fractures were associated with an increased incidence of cardiorespiratory diseases among adults with intellectual disabilities.

Child oral health and preventive dental service access among children with intellectual disabilities, autism and other educational additional support needs: A population-based record linkage cohort study.

OBJECTIVE: Inequalities in child oral health are a global challenge and the intersection of socioeconomic factors with educational additional support needs (ASN), including children with intellectual disabilities or autism, have thus far received limited attention in relatively small clinical studies. We aimed to address this evidence gap by investigating oral health and access to preventive dental services among children with ASN compared to the general child population. METHODS: Cohort study linking data from six Scotland-wide health and education databases compared: dental caries experience and tooth extraction via general anaesthetic; receipt of school-based dental inspection; access to primary care and hospital dental services; and access to the Childsmile national oral health improvement programme between children with a range of ASN (intellectual disabilities, autism, social and other) and their peers for the school years 2016/17-2018/19 (n = 166 781). RESULTS: Children with any ASN had higher rates of caries experience than those with no ASN, however, after adjustment for socioeconomic deprivation, sex, year, and school type only those with a social or other ASN remained at increased risk. Rates of tooth extraction under general anaesthesia in hospital were higher among children with intellectual disabilities (aRR = 1.67;95% CI = [1.16-2.37]). School-based dental inspection access improved for children with intellectual disability and/or autism from 2016/17 onwards, although higher rates of child refusal on the day were observed in these groups (no ASN refusal: 5.4%; intellectual disability: 35.8%; autism: 40.3%). Children with any ASN were less likely to attend primary dental-care regularly, and in those who attended, children with intellectual disability or autism were less likely than their peers to receive prevention (fluoride varnish, oral-hygiene instruction, or dietary advice). Childsmile nursery-supervised toothbrushing programme access among children with any ASN was similar to children with no ASN and children with intellectual disability (aRR = 1.27;95% CI = [1.12-1.45]) or autism (aRR = 1.32;95% CI = [1.19-1.45]) were more likely to receive support from Childsmile dental health support worker. CONCLUSIONS: We have identified inequalities in oral health and dental care for children with different ASN in Scotland with both a greater burden of disease among some groups and higher complexity of care; compounded by reduced and variable access to preventive dental services. Further efforts are needed to develop and improve preventive care pathways for children with ASN and integrate oral health to wider healthcare systems for these children to mitigate against oral health inequalities.

Utilising Interview Methodology to Inform the Development of New Clinical Assessment Tools for Anxiety in Autistic Individuals Who Speak Few or no Words.

Autistic individuals with intellectual disability who speak few or no words are at high risk of anxiety but are underrepresented in research. This study aimed to describe the presentation of anxiety in this population and discuss implications for the development of assessments. Interviews were conducted with 21 parents/carers of autistic individuals and nine clinicians. Data were analysed using content analysis and interpretative phenomenological analysis. Anxiety behaviours described by parents/carers included increased vocalisation, avoidance and behaviours that challenge. Changes to routine were highlighted as triggering anxiety. Clinicians discussed the importance of identifying an individual's baseline of behaviour, knowing an individual well and ruling out other forms of distress. This study raises considerations for early identification of anxiety and for subsequent support.

Assessment of Trabecular Bone Score: a 7-year follow-up study in institutionalized adults with refractory epilepsy and intellectual disability.

PURPOSE: The aim of this longitudinal study was to assess trabecular bone scores (TBS) in institutionalized adults with refractory epilepsy and intellectual disability and to study the association of TBS and incident fractures during seven years of follow-up. METHODS: In 2009 and 2016, all institutionalized adult patients of a long-stay care facility in the Netherlands (n=261) were invited to undergo a dual-energy X-ray absorptiometry (DXA) including vertebral fracture assessment (VFA) and assessment of TBS. Vertebrae T4-L4 were analyzed using quantitative morphometry. New and worsening vertebral fractures (VFs) were considered as incident VFs. Data regarding clinical fractures were extracted from the medical files. Patients were treated with anti-osteoporosis medication according to the Dutch guideline. RESULTS: Baseline and follow-up DXA, VFA and TBS could be obtained in 136 patients (83 male) aged between 18 and 79 years old (44.7±15.5). At baseline, 36 patients (26.5%) were diagnosed with osteoporosis, 68 (50.0%) with osteopenia and 32 patients (23.5%) had a normal bone mineral density (BMD). As for TBS, 26 patients (19.1%) had a partially degraded microarchitecture and 26 patients (19.1%) a degraded microarchitecture. During seven years of follow-up, 80 patients (59%) sustained at least one fracture, of which 28 patients (35%) had one or more major osteoporotic fractures. Thirty-four patients (25.0%) had at least one new or worsening morphometric VF. Compared to baseline, TBS significantly decreased over seven years of follow-up in non-treated patients (-0.039±0.064, p<.001). In patients who were treated with bisphosphonates for more than one year during follow-up, TBS did not change significantly (p=.093). In multivariate analyses, no significant associations were found between TBS at baseline and incident fractures during follow-up. CONCLUSION: In this study, we found a high incidence of fractures and TBS decreased significantly over seven years of follow-up in non-treated institutionalized adult patients with refractory epilepsy and intellectual disability, but TBS was not associated with incident fractures.

Fertility rates in women with intellectual and developmental disabilities in Wisconsin Medicaid.

BACKGROUND: Women with intellectual and developmental disabilities (IDD) face stigma and inequity surrounding opportunity and care during pregnancy. Little work has quantified fertility rates among women with IDD which prevents proper allocation of care. OBJECTIVE: Our objective was to cross-sectionally describe fertility patterns among women with and without intellectual and developmental disabilities (IDD) in 10-years of Medicaid-linked birth records. STUDY DESIGN: Our sample was Medicaid-enrolled women with live births in Wisconsin from 2007 to 2016. We identified IDD through prepregnancy Medicaid claims. We calculated general fertility-, age-specific-, and the total fertility-rates and 95% confidence intervals (95% CI) for women with and without IDD and generated estimates by year and IDD-type. RESULTS: General fertility rate in women with IDD was 62.1 births per 1000 women with IDD (95% CI 59.2, 64.9 per 1000 women) and 77.1 per 1000 for women without IDD (95% CI: 76.8, 77.4 per 1000 women). General fertility rate ratio was 0.81 (95% CI: 0.7, 0.9). Total fertility was 1.80 births per woman with IDD and 2.05 births per woman without IDD (rate ratio: 0.89 95% CI: 0.5, 1.5). Peak fertility occurred later for autistic women (30-34 years), compared with women with other IDD (20-24 years). CONCLUSION: In Wisconsin Medicaid, general fertility rate of women with IDD was lower than women without IDD: the difference was attenuated when accounting for differing age distributions. Results highlight the disparities women with IDD face and the importance of allocating pregnancy care within Medicaid.

Use of the Griffiths mental development scale-Chinese in the assessment of children with autism spectrum disorder and global developmental delay/intellectual disability.

ABSTRACT: The Griffiths Mental Development Scale-Chinese (GDS-C) is used in China to assess the development of children from birth to 8 years of age. Language disorders are a common symptom of autism spectrum disorder (ASD) and global developmental delay (GDD)/intellectual disability (ID). There is a need to identify distinct clinical characteristics in children suspected of having these 2 disorders, mainly presenting as language disorders. Here, we aimed to use the GDS-C to evaluate children presenting with language problems to identify characteristics that distinguish ASD and GDD/ID. Children with language problems were recruited between August 2018 and December 2019. A total of 150 children aged 25 to 95.2 months were enrolled (50 in the ASD group, 50 in the GDD/ID group, and 50 in the typical group). Each group was subdivided by age as follows: 24-36 months, >36-60 months, and >60-96 months. Developmental characteristics assessed using the GDS-C were analyzed and compared. Both, children with ASD and GDD/ID presented with a lower developmental level than typical children in all six subscales of the GDS-C. No significant differences were observed in the six subscale scores between the ASD and GDD/ID groups, except for the practical reasoning subscale score in the >36 to 60 months subgroups, which was significantly lower in the GDD/ID group than in the ASD group. The developmental imbalance of subscales within the ASD and GDD/ID groups identified troughs in the personal-social, language, and practical reasoning areas in children with ASD and in the language and practical reasoning areas in children with GDD/ID relative to typical children. The GDS-C is a useful, comprehensive tool for the assessment of the developmental state of children with ASD and GDD/ID. Characteristics of practical reasoning subscale help diagnose autism in >36 to 60 months old children.

The phenotype of 15 cases with rare 8q24.13-q24.3 deletions-A new syndrome or still an enigma?

Diagnosis of rare copy number variants (CNVs) with scarce literature evidence poses a major challenge for interpretation of the clinical significance of chromosomal microarray analysis (CMA) results, especially in the prenatal setting. Bioinformatic tools can be used to assist in this issue; however, this prediction can be imprecise. Our objective was to describe the phenotype of the rare copy number losses encompassing the 8q24.13-q24.3 locus, and to find common features in terms of genomic coordinates, gene content, and clinical phenotypic characteristics. Appropriate cases were retrieved using local databases of two largest Israeli centers performing CMA analysis. In addition, literature and public databases search was performed. Local database search yielded seven new patients with del (8)(q24.13q24.3) (one of these with an additional copy number variant). Literature and public databases search yielded eight additional patients. The cases showed high phenotypic variability, ranging from asymptomatic adults and fetuses with normal ultrasound to patients with autism/developmental delay (6/11 postnatal cases, 54.5%). No clear association was noted between the specific disease-causing/high-pLI gene content of the described del (8)(q24.13q24.3) to neurodevelopmental disorders, except for a possibly relevant locus encompassing the KCNQ3 gene. We present the challenges in classification of rare variants with limited clinical information. In such cases, genotype-phenotype correlation must be assessed with extra-caution and possibly using additional methods to assist the classification, especially in the prenatal setting.

COVID-19 outcomes among people with intellectual and developmental disability in California: The importance of type of residence and skilled nursing care needs.

BACKGROUND: People with intellectual and developmental disabilities (IDD) appear to be at greater risk for severe outcomes from COVID-19. The roles of congregate living and skilled nursing care needs in this disparity are unclear. OBJECTIVE: To determine the impact of residential setting and level of skilled nursing care on COVID-19 outcomes for people receiving IDD services, compared to those not receiving IDD services. METHODS: Utilizing publicly available California data on COVID-19 outcomes for people receiving IDD services (early May through October 2, 2020), we report outcomes based on seven types of residence, differentiated by number of residents and level of skilled nursing care provided. We compared these results to the larger California published outcomes. RESULTS: Compared to Californians not receiving IDD services, in general, those receiving IDD services had a 60% lower case rate, but 2.8 times higher case-fatality rate. COVID-19 outcomes varied significantly among Californians receiving IDD services by type of residence and skilled nursing care needs: higher rates of diagnosis in settings with larger number of residents, higher case-fatality and mortality rates in settings that provided 24-h skilled nursing care. CONCLUSIONS: Diagnosis with COVID-19 among Californians receiving IDD services appears to be related to the number of individuals within the residence, while adverse COVID-19 outcomes were associated with level of skilled nursing care. When data is available, future research should examine whether these relationships persist even when controlling for age and pre-existing conditions.

Pregnancy complications and maternal birth outcomes in women with intellectual and developmental disabilities in Wisconsin Medicaid.

BACKGROUND: Women with intellectual and developmental disabilities (IDD) may face greater risk for poor pregnancy outcomes. Our objective was to examine risk of maternal pregnancy complications and birth outcomes in women with IDD compared to women without IDD in Wisconsin Medicaid, from 2007-2016. METHODS: Data were from the Big Data for Little Kids project, a data linkage that creates an administrative data based cohort of mothers and children in Wisconsin. Women with ≥1 IDD claim the year before delivery were classified as having IDD. Common pregnancy complications and maternal birth outcomes were identified from the birth record. We calculated risk ratios (RR) using log-linear regression clustered by mother. We examined outcomes grouped by IDD-type and explored interaction by race. RESULTS: Of 177,691 women with live births, 1,032 (0.58%) had an IDD claim. Of 274,865 deliveries, 1,757 were to mothers with IDD (0.64%). Women with IDD were at greater risk for gestational diabetes (RR: 1.28, 95% CI: 1.0, 1.6), gestational hypertension (RR: 1.22, 95% CI: 1.0, 1.5), and caesarean delivery (RR 1.32, 95% CI: 1.2, 1.4) compared to other women. Adjustment for demographic covariates did not change estimates. Women with intellectual disability were at highest risk of gestational hypertension. Black women with IDD were at higher risk of gestational hypertension than expected under a multiplicative model. CONCLUSIONS: Women with IDD have increased risk of pregnancy complications and adverse outcomes in Wisconsin Medicaid. Results were robust to adjustment. Unique patterns by IDD types and Black race warrant further exploration.

Creating equitable healthcare quality and safety for children with intellectual disability in hospital.

Children with intellectual disability are susceptible to poor experiences of care and treatment outcomes, and this may compound existing health inequities. Evidence to date indicates three priority areas that must be addressed in order to reduce these inequities in the safety and quality of care for children with intellectual disability. Firstly, we need reliable methods to identify children with intellectual disability so that healthcare organizations understand their needs. Secondly, we need to develop quality metrics that can assess care quality and unwarranted care variation for children with intellectual disability in hospital. Finally, for a comprehensive understanding of the safety and quality of care for these children, and how to improve, it is critical that healthcare organizations partner with parents/carers and enable children with intellectual disability to voice their experiences of care. Children with intellectual disability have higher healthcare utilization than their peers; yet, their voice is rarely sought to optimize the safety and quality of their healthcare experience. Patient experience narratives enhance our understanding of the genesis of adverse events. By addressing these priorities, children with intellectual disability will be identified, and health services will measure and understand the problematic and beneficial variations in care delivery and can then effectively partner with children and their parents/carers to address the inequities in care quality and create safer healthcare.

Improving the participation of adults with visual and severe or profound intellectual disabilities: a process evaluation of a new intervention.

BACKGROUND: While the participation of adults with visual and severe or profound intellectual disabilities (VSPID) in society and community life is important, evidence-based interventions to improve their participation are lacking. We conducted a process evaluation of the implementation of 'Care for Participation+' (CFP+), a new intervention targeting the attitudes of direct support professionals (DSPs) toward the participation of adults with VSPID, within a residential facility in the Netherlands. METHODS: CFP+ was inspired by the Boston Psychiatric Rehabilitation Approach and adapted by adopting a new definition and operationalization of the concept of participation for adults with VSPID. Following systematic training, 16 DSPs of adults with VSPID were able to apply key elements of CFP+ to explore diverse roles and activities for this population, facilitating their self-management, teaching them necessary skills for participation, and organizing support. Our process evaluation entailed an investigation of the delivered dose, reach, fidelity, and adaptation of CFP+ during and after the CFP+ intervention. We also evaluated the mechanisms of impact and context using questionnaires, assignments, documentation, interviews, and a logbook. RESULTS: The intended dose, reach, and fidelity relating to the implementation of CFP+ were not achieved. Despite this fact, an assessment of the mechanisms of impact indicated that assignments of CFP+ were well (75%) or reasonably well (17%) understood by DSPs. CFP+ was applied by DSPs to stimulate self-management (83% of DSPs), new activities (100%), enhanced involvement in existing activities (67%) and to explore new roles (50%) for adults with VSPID. A negative contextual factor mentioned by the trainer and manager was the DSPs' lack of commitment to the training program. Another negative contextual factor mentioned by DSPs was the lack of time for implementing CFP+. CONCLUSIONS: CFP+ provides new opportunities to improve the participation of adults with VSPID. Despite the non-optimal conditions for implementing CFP+ and the DSPs' general reluctance to apply the new intervention, some have actively used CFP+ within the residential facility. Future studies should focus on the outcomes of CFP+ regarding attitudinal changes among DSPs relating to the participation of adults with VSPID and their quality of life.

Toward Actionable Practice Parameters for "Dual Diagnosis": Principles of Assessment and Management for Co-Occurring Psychiatric and Intellectual/Developmental Disability.

PURPOSE OF REVIEW: Although treatment algorithms and parameters for best practice are readily available for all major syndromes of psychiatric impairment, the occurrence of psychiatric syndromes in individuals with intellectual and developmental disability (IDD) invokes serious contextual challenges for interpretation of symptoms, diagnosis, and optimization of treatment, both for clinicians and for the service sectors in which care and support of individuals with IDD are delivered. Recognizing that there exist very few definitive resources for best practice under the circumstance of this form of "dual diagnosis," the Missouri Department of Mental Health convened an expert panel to conduct a focused review and synthesis of the relevant scientific literature from which to develop guidance in the form of decision support to clinicians. This article summarizes the findings for three of the most common and impairing clusters of psychiatric symptoms that co-occur with IDD-aggression, depression, and addictions. RECENT FINDINGS: Individuals with IDD are at high risk for the development of psychiatric symptoms (PS), which often manifest uniquely in IDD and for which evidence for effective intervention is steadily accruing. Interventions that are commonly implemented in the IDD service sector (e.g., functional communication training and positive behavioral support planning) are capable of mitigating severe behavioral impairment, yet rarely invoked when dual diagnosis patients are seen in the psychiatric service sector. Conversely, state-of-the-art interventions for traumatic stress, pharmacotherapy, and psychotherapy have proven capable of improving behavioral impairments in IDD but are typically restricted to the psychiatric service sector, where there exist significant barriers to access for patients with IDD, including limitations imposed by diagnostic eligibility and practitioner experience. Bridging these gaps in knowledge and clinical capacity across the respective IDD and PS service sectors should be of very high priority in strategizing the care and support of IDD patients with serious co-occurring psychiatric conditions.

Scoping review protocol documenting cancer outcomes and inequalities for adults living with intellectual and/or developmental disabilities.

INTRODUCTION: There is increasing attention on the cancer burden for adults with intellectual and developmental disabilities (IDD). Emerging evidence suggests there are differences in cancer experiences and outcomes for individuals living with IDD, from risk through survivorship. These differences may be attributed to features of the IDD, such as cognitive deficits and communication, as well as social determinants of health-like lower education levels and ableism. However, there is no comprehensive overview of the literature quantifying these potential disparities and describing the influencing factors. In this paper, we describe a scoping review protocol to systematically review published literature on cancer for adults with IDD. The purpose of this review is to identify differences in cancer risk, stage at diagnosis, treatment and survival along the cancer continuum for adults with IDD and outline potential contributing factors creating these disparities. METHODS AND ANALYSIS: We will follow Arksey and O'Malley's expanded framework for scoping reviews to conduct this review. We will systematically search electronic databases for peer-reviewed, published journal articles to identify appropriate studies in collaboration with a health science librarian. Two reviewers will independently review titles and abstracts followed by a full-text review to determine whether it meets inclusion criteria. A data chart for collecting and sorting information will be developed in consultation with the team. Results will be collated and reported following the Preferred Reporting Items for Systematic Reviews and Meta-Analyses (PRISMA) extension for scoping reviews: PRISMA-Scoping Reviews. Extracted information will be summarised quantitatively and qualitatively to meet review objectives. ETHICS AND DISSEMINATION: This scoping review will employ a methodology to identify literature related to cancer outcomes and experiences for adults with IDD. Results will be disseminated to relevant stakeholders who care for and support individuals with IDD at local, provincial and national levels and through publishing findings. By highlighting the disparities in the cancer system and gaps in the research, this scoping review can provide direction for future action.

Quantitative gait assessment in children with 16p11.2 syndrome.

BACKGROUND: Neurodevelopmental disorders such as 16p11.2 syndrome are frequently associated with motor impairments including locomotion. The lack of precise measures of gait, combined with the challenges inherent in studying children with neurodevelopmental disorders, hinders quantitative motor assessments. Gait and balance are quantifiable measures that may help to refine the motor phenotype in 16p11.2. The characterization of motor profile is useful to study the trajectories of locomotion performance of children with genetic variants and may provide insights into neural pathway dysfunction based on genotype/phenotype model. METHODS: Thirty-six children (21 probands with 16p11.2 deletion and duplication mutation and 15 unaffected siblings), with a mean age of 8.5 years (range 3.2-15.4) and 55% male, were enrolled. Of the probands, 23% (n = 6) had a confirmed diagnosis of autism spectrum disorder (ASD) and were all male. Gait assessments included 6-min walk test (6MWT), 10-m walk/run test (10MWR), timed-up-and-go test (TUG), and spatio-temporal measurements of preferred- and fast-paced walking. The Pediatric Evaluation of Disability Inventory-Computer Adaptive Tests (PEDI-CAT), a caregiver-reported functional assessment, was administered. Measures of balance were calculated using percent time in double support and base of support. Analyses of the six children with ASD were described separately. RESULTS: Thirty-six participants completed the protocol. Compared with sibling controls, probands had significantly lower scores on the 6MWT (p = 0.04), 10MWR (p = 0.01), and TUG (p = 0.005). Group differences were also identified in base of support (p = 0.003). Probands had significantly lower PEDI-CAT scores in all domains including the mobility scale (p < 0.001). Using age-matched subsamples, the ASD and non-ASD genetic variant groups had larger base of support compared to the controls. In the fast-paced condition, all participants increased their velocity, and there was a corresponding decrease in percent time in double support compared to the preferred-pace condition in all participants. Only the ASD group presented with upper limb arm/hand stereotypies. CONCLUSIONS: Children with 16p11.2, with and without ASD, present with balance impairment during locomotion activities. Probands performed worse on functional assessments, and quantitative measures revealed differences in base of support. These results highlight the importance of using precise measures to differentiate motor dysfunction in children with neurodevelopmental disorders.

Syntactic development in children with intellectual disabilities - using structured assessment of syntax.

BACKGROUND: Children with intellectual disabilities (IDs) have a severe delay in syntactic development compared with other language abilities. This study investigated conditions of syntactic development in native Japanese-speaking children with ID. METHODS: Children with ID [N = 51; 18 autism spectrum disorders (ASD), 18 Down syndrome (DS) and 15 ID without ASD and DS] were compared with typically developing children (N = 78) with the same mental age (MA). The development of syntax in spoken language was examined by receptive and production tasks. RESULTS: The development of syntax in children with ID was significantly delayed than in typically developing children with the same MA. However, when reaching the MA of 7-9, syntax abilities started to develop remarkably. Moreover, children with ASD had significant difficulties in acquiring passive voice, whereas children with DS showed a significant delay in syntactic development. CONCLUSIONS: The development of syntax in children with ID might be affected by MA and the type of disability. Moreover, it is necessary to exceed an MA of 7-9 years for children with ID to develop syntax abilities.