ABSTRACT

Background

Intellectual and developmental disabilities (IDD) are varied in their nature and presentation. Barriers to oral healthcare are reported in studies of general populations with IDD but these may not reflect the barriers experienced by individuals with rare disorders such as Rett syndrome (RTT). There are also few peer-reviewed studies in the Australian context exploring barriers to dental care access for patients living with a disability. This qualitative study explored caregivers' perceptions and experiences regarding oral health and access to dental care for those with RTT in Australia.

Methods

Parents of 31 individuals with a confirmed MECP2 mutation were sampled purposively from the Australian Rett Syndrome Database. Interview questions were based on earlier studies used in other disability populations and queried identification and management of dental pain and influence of other comorbidities in their child's oral care. Interviews were audio-recorded, transcribed and analysed using NVivo (Version 12 Plus). Directed content analysis was used to code data to a framework constructed from a literature review of factors affecting access to professional oral healthcare systems and factors affecting access to optimal at-home oral care in disability.

Results

The most frequently cited barriers to professional dental care were dentist-related, while caregiver related financial barriers were cited by a minority of families. Dentist-related financial barriers were not present in these data. Most factors affecting access to optimal at-home oral care coded to the existing framework, with further enablers identified under training for the caregiver or parent.

Conclusions

The findings of this study provide a point of reference to understand factors affecting provision of at-home dental care and professional services to enable optimal oral health in RTT. Future research could explore the provision of targeted oral health information on RTT to carers and clinicians.

No abstract is available for this article.

ABSTRACT

Background

Health care accessibility and inequality for people with intellectual disabilities have long been subjects of discussion. Issues related to eye examinations and visits to ophthalmologists are particularly challenging for these people. However, disability studies and healthcare from the perspectives of people with intellectual disabilities remain scarce. The current study aims to explore the experiences and expectations of adults with intellectual disabilities during ophthalmologist visits, with a focus on their subjective viewpoints.

Methods

A qualitative study was conducted between 2021 and 2022 that involved semistructured interviews with 22 adults with intellectual disabilities who were users of day programs or residential services in Taiwan. The interviews, supplemented with pictures, were conducted using purposive sampling. Thematic analysis was used to analyse the data.

Findings

Four themes emerged regarding their experiences with ophthalmologists: ‘Because my eyes itched, I went to the doctor;’ the need for clear and slow explanations during vision screening; anxiety and discomfort during equipment use and focused examination; and ‘The doctor is good. She smiles and is gentle with me.’ In addition, two themes related to their expectations were identified:equipment use and pre-examination instructions and ideal characteristics of ophthalmologists: smiling and talking slowly.

Conclusion

For people with intellectual disabilities, an ideal ophthalmologist is patient-centred and provides individualised care—‘Smiling and Talking Slowly’; otherwise, fear of visiting eye doctors worsens their health inequalities. Integrating these considerations into ophthalmology training and practice is essential to enhance the quality of care for and the well-being and dignity of people with intellectual disabilities.

ABSTRACT

Background

Individuals with PWS need constant support and/or supervision, which creates a high caregiver burden on their parents and siblings. Previous research has identified adverse stress outcomes in relatively small and country-specific samples. This study's aims were to examine stress outcomes in a large multi-country sample of parents and siblings and to expand upon previous research by incorporating data on psychosocial factors that may mitigate stress outcomes.

Methods

The sample comprised 135 parents of a child with PWS, with additional data for 45 siblings as reported by parents. Participants were recruited from 31 countries, spanning Europe, North and South America, Africa, Asia and Australasia, who participated by completing an online questionnaire that included standardised psychometric measures of depression and anxiety (HADS), life stress (PSS), PTSD symptoms (CATS-C) and family cohesion (FACES II). Outcomes were compared to published population norms, and multiple regression was used to investigate the role of potential exacerbating and mitigating factors.

Results

Findings revealed high rates of mental pathologies in both parents and siblings. Parents' scores for depression and anxiety indicated high rates of caseness: 67.4% of parents exhibited ‘abnormal’ levels of anxiety, while 15.6% exhibited ‘borderline abnormal’ levels; 34.8% exhibited ‘abnormal’ levels of depression, with 22.2% exhibiting ‘borderline abnormal’ levels. Younger parents exhibited higher anxiety than older parents (p = 0.007); younger male parents reported higher depression than older male parents (p = 0.029). Parents whose child with PWS lived in the family home exhibited higher depression scores than parents whose child with PWS lived away from home (p = 0.035). Family cohesion was inversely associated with parental depression (p < 0.001) and parental anxiety (p = 0.012), even when statistically controlling for age of parent, age of child with PWS and parental education level. Scores for life stress were markedly higher than population norms, with 88.7% of parents exhibiting ‘high’ or ‘moderate’ life stress. Parental life stress was significantly correlated with temper outburst severity in their child with PWS (p < 0.001) and with food problem severity (p < 0.001). All siblings exhibited at least one symptom of PTSD, with 28.9% of siblings exhibiting ‘clinically relevant’ levels of PTSD symptoms. Sibling PTSD symptom levels were significantly associated with temper outburst severity in the child with PWS (p = 0.025) but not with ratings of food problem severity (p = 0.114). Family cohesion was inversely associated with PTSD symptoms in siblings (p = 0.022).

Conclusions

PWS impacts families negatively, and relatives suffer as a result. The findings of this study confirm that parents and siblings of persons with PWS exhibit clinically notable levels of mental pathology. Strategies to enhance family cohesion should be employed to help diminish adverse outcomes among PWS families.

ABSTRACT

Background

Although sexual health literacy is recognised as critical to sexual health and well-being, little is known about how people with intellectual disabilities acquire or use sexual health literacy skills. This scoping review examined research to explore what is known about sexual health literacy among adults with intellectual disability.

Method

We used Arksey and O′Malley's scoping framework to guide our review. We searched five electronic databases and reference lists of full-text articles. Inclusion criteria included (i) original research in peer reviewed journals; (ii) published in English; (iii) addressed perspectives or experiences of people with intellectual disabilities regarding sexual health literacy or related topics. Findings were reported according to the Preferred Reporting Items for Systematic Reviews and Meta-analyses extension for scoping reviews (PRISMA-Scr). All text labelled ‘results’, ‘findings’ or ‘discussion’ was subjected to interpretive content analysis.

Results

The search strategy identified 5447 records, 102 met all eligibility criteria and were included for review. A conceptual framework to understand factors shaping sexual health literacy was developed. In this framework, sexual health literacy is underpinned by two fundamental pillars: sexual knowledge and sexual autonomy. The availability of sexual knowledge and sexual autonomy is threatened by several factors in the social environment: presumed sexual vulnerability and sexual incompetence, and expectations of heteronormativity and asexuality. Combined these factors reduce opportunities for acquisition of sexual knowledge and enactment of sexual autonomy and ultimately appear to limit sexual literacy among people with intellectual disabilities.

Conclusion

Sexual health literacy is critical for people with intellectual disabilities to enjoy full sexual citizenship. Despite this limited attention has been paid to supporting people with intellectual disabilities to acquire and use sexual health literacy skills. Comprehensive education programs are needed to address this gap and ensure the rights of people with intellectual disabilities to engage in safe, pleasurable sexual experiences and enjoy good sexual health are upheld.

ABSTRACT

Over time, sexuality has become a crucial aspect in people's lives, regardless of physical, intellectual or social conditions, ranging from sex and sexually transmitted diseases to gender identity and sexual orientation. The aim of this study was to carry out a review of the scientific literature on sexual orientation in people with intellectual disabilities from the Education and Health Sciences and to analyse how sex education is addressed in this group. To this end, a systematic review of research on sexual orientation in people with intellectual disabilities was carried out. The results obtained reveal that there is hardly any scientific corpus on this subject, so emphasis should be placed on the attitudes towards LGTBIQA+ people with intellectual disabilities, as well as on the training and understanding of the different sexual orientations they may have. It is essential to face the obstacles, stigmas and prejudices established in society to normalise the sexual needs of these people, improving, consequently, their quality of life.

ABSTRACT

Background

Borderline intellectual functioning (BIF) is a condition that involves limitations in cognitive skills that affect the daily functioning of people who present it. BIF has lost visibility in diagnostic manuals such as the DSM-5-TR and ICD-11, which relegate it to complementary categories of aspects requiring attention, rather than recognising it as a category of its own. This lack of recognition as well as unclear criteria for diagnosis underscores the need for a deeper understanding of BIF.

Method

The study conducted a scoping review to map the available evidence in the field of BIF. Following the PRISMA-ScR framework, ProQuest, WoS, SCOPUS and EBSCOhost databases were searched. Documents were selected based on inclusion criteria: date of publication (since 2012), study of BIF as a specific group and language (English or Spanish). A total of 138 documents were included, both academic and grey literature.

Results

The review mapped the literature into key categories: intellectual functioning, adaptive functioning and additions for a comprehensive evaluation. Most research focused on comorbid psychiatric, emotional and behavioural disorders associated with BIF, as well as cognitive aspects. Very few addressed adaptive functioning explicitly, a crucial area for diagnosing and supporting individuals with BIF, though many covered its domains (conceptual, social and practical). Studies predominantly used quantitative methodologies, with only a few incorporating qualitative methods and directly involving people with BIF.

Conclusions

The review emphasises the need for a clearer definition of BIF. Future studies should incorporate the perspectives of individuals with BIF to fully understand their needs and challenges across various life domains.

ABSTRACT

Background

Dravet Syndrome (DS), Helsmoortel-Van Der Aa Syndrome (HVDAS) and Tuberous Sclerosis Complex (TSC) are rare genetic syndromes, sharing intellectual disability (ID) and motor delay. In DS, two distinct gait patterns, crouch and non-crouch, have been described using instrumented 3D gait analysis (i3DGA). This cross-sectional study measures gait in participants with TSC and HVDAS. The findings are compared to the known crouch and non-crouch gait patterns observed in DS and to typical gait.

Methods

Participants (6–22 years) with DS (n = 37; 19 crouch and 18 non-crouch), HVDAS (n = 12) or TSC (n = 8) were compared with typically developing (TD) peers (n = 33). All participants underwent i3DGA (Plugin Gait model processed with Vicon Nexus and MATLAB®) to investigate spatiotemporal and lower-limb kinematics.

Results

All three genetic syndromes showed increased step width. Participants with HVDAS and DS, but not participants with TSC walked with decreased step length and velocity compared to TD. HVDAS demonstrated increased knee flexion during the stance phase, lack of hip extension during pre-swing, and increased ankle dorsiflexion during some phases of the gait cycle (p < 0.001). Additionally, HVDAS showed similar kinematic deviations to DS-NonCrouch. No significant differences were found in terms of kinematics between TSC and TD peers (p > 0.05).

Conclusion

The current study reveals differences in gait characteristics from typical functional gait in rare genetic disorders. DS-Crouch, DS-NonCrouch and HVDAS display a more impaired gait from a biomechanical perspective than TSC. The variability of clinical and genetic features might explain heterogeneity in gait deviations and should be further explored.

ABSTRACT

Background

Adults with intellectual and/or developmental disabilities are disproportionately excluded from participation in research, consequently increasing disparities experienced by this group. Gatekeepers, individuals who control access to research participation opportunities, may either support or pose a barrier to inclusion in research. We sought to understand how gatekeepers serve as supports and barriers to the participation of people with intellectual and/or developmental disabilities in research and the factors that may drive their actions. We also sought to identify approaches to interacting with gatekeepers that promoted the sharing of research participation opportunities.

Methods

We conducted a scoping review of manuscripts published between January 2009 and September 2024 describing gatekeeping during recruitment for adults with intellectual and/or developmental disabilities in social/behavioural research. We conducted content analysis of 22 manuscripts to identify ‘gate opening’ actions (actions that support research participation) and ‘gate closing’ actions (actions that pose a barrier to research participation) at the point of recruitment. We also identified approaches researchers took when interacting with gatekeepers to promote gate opening actions.

Results

Gatekeepers' attitudes (e.g., valuing research) and knowledge about prospective participants were associated with gate opening actions. Gatekeepers' attitudes of mistrust of researchers and/or research, deprioritisation of research and presumed incapacity of people with intellectual and/or developmental disabilities to consent to, participate in and/or benefit from research participation were associated with gate closing actions. Gatekeepers' lack of information (e.g., about research and prospective participants) was also associated with gate closing actions. Restrictive organisational policies and gatekeepers' lack of resources (e.g., time) were also associated with gate closing actions. Approaches for interacting with gatekeepers that may foster gate opening actions included: addressing gatekeeper concerns, educating gatekeepers about the benefits of research participation and developing relationships with gatekeepers.

Conclusion

We identified several malleable factors that may drive gate opening and gate closing actions. Enhanced collaboration between researchers and gatekeepers may foster greater opportunities for individuals with intellectual and/or developmental disabilities to learn about research opportunities.

ABSTRACT

Background

The connection between sleep problems and behavioural challenges in individuals with Down syndrome (DS) has been a central focus of research across different age groups. However, uncertainty remains regarding this association, particularly when relying solely on daily-based assessments. This study aimed to examine the relationship between sleep problems and behavioural challenges at both overall (macro) and daily (micro) levels among youth with DS.

Methods

Sleep disturbances were measured in a sample of 65 youth with DS aged 6–17 years using the Children's Sleep Habits Questionnaire (CSHQ) and actigraph watches assessing sleep efficiency, sleep duration and wake after sleep onset. Behavioural challenges were evaluated through externalising and internalising subscales of the Child Behavior Checklist (CBCL) and of the Scales of Independent Behavior, Revised (SIB-R).

Results

The findings demonstrated that over a period of time, sleep problems are significantly associated with both externalising and internalising behaviours as measured by CSHQ and CBCL, even after accounting for the effects of IQ and SIB-R Broad Independence. No significant correlations were observed on a daily basis over seven consecutive days, as measured by actigraphy and both externalising and internalising indices of SIB-R.

Conclusions

The results highlight the complexity of the sleep–behaviour relationship in DS, indicating that while chronic sleep issues impact long-term behaviours, nightly variations do not predict immediate behavioural changes.

ABSTRACT

Background

Adults with Down syndrome (DS) experience an increased risk of Alzheimer's disease (AD). Valid cognitive assessments for adults with DS with severe/profound intellectual disability (ID) are needed. It is unclear whether eye tracking is feasible for detecting AD in DS.

Method

Fifty-three adults with DS completed a visual paired comparison (VPC) task, a battery of cognitive measures, and underwent PET scanning. Study partners reported on the participant's dementia symptoms. Bivariate correlations assessed associations between eye-tracking metrics and AD-related pathology and symptomatology. Analyses included the full sample (n = 53) and a subgroup with an IQ ≤ 45 (n = 33).

Results

Greater fixation duration during the habituation phase was associated with better cognitive performance on the Modified Cued Recall Test (mCRT) (intrusions: r = −0.39, p = 0.011) and less PET tau (r = −0.47, p = 0.014). Larger saccadic amplitudes during the test phase were associated with younger age (r = −0.45, p < 0.001), better cognitive performance on the mCRT (total: r = 0.31, p = 0.041; intrusions: r = −0.33, p = 0.032) and less PET Aβ (r = −0.40, p = 0.025). Greater preference to fixate on the novel image was associated with fewer dementia symptoms (count: r = −0.44, p = 0.002; duration: r = −0.38, p = 0.009). This pattern of significance remained for the subgroup with lower IQ scores.

Conclusion

The VPC task is a potentially useful method for assessing AD-related cognitive impairments in adults with DS across varying ID levels.

ABSTRACT

Background

Resting metabolic rate (RMR), the energy required by the body at rest, is the largest part of total daily energy expenditure. Commonly used prediction equations may overestimate RMR in adults with Down syndrome (DS). The purpose of this study was to assess the equivalency of prediction equations for estimating RMR in adults with DS.

Methods

Twenty-five adults with DS (24 ± 5 years of age, 64% female) completed RMR assessments at an academic medical centre in the United States between November 2021 and July 2023. Measured RMR (kilocalories per day) was compared to estimated RMR from eight prediction equations using a null hypothesis significance (i.e., a paired t-test) and equivalence (i.e., a two one-sided test) tests. Bland–Altman plots, Pearson correlations and linear regressions were used to evaluate the bias between the measured and predicted RMR values.

Results

Measured RMR in adults with DS was 1090 ± 136 kcal/day. Prediction equations overestimated RMR by 8 ± 16% (76 ± 165 kcal/day) to 45 ± 16% (488 ± 165 kcal/day) except for the Bernstein fat-free mass equation which underestimated RMR by 0.2 ± 11.5% (8 ± 123 kcal/day) and was statistically equivalent to measured RMR in our sample (p = 0.027).

Conclusions

The Bernstein fat-free mass equation offers better accuracy in adults with DS than other RMR prediction equations, but the equation needs to be evaluated in larger, more diverse samples of adults with DS.

ABSTRACT

Background

Adults with intellectual disability (ID) experience injurious falls that may affect their quality of life. The present randomised control trial (RCT) study examined the efficacy of a 10-week proprioceptive training programme, on static balance performance, in adults with mild to moderate ID.

Methods

Participants were voluntarily recruited from a day care centre and randomly assigned into the intervention (IG; n = 14; 36.4 ± 3.8 years; males/females = 8/6) and the control group (CG; n = 13; 37.6 ± 4.8 years; males/females = 7/6). The IG trained three times per week with the proprioceptive training programme, whereas both groups followed the regular adapted physical activity programme of the day care centre. Static balance was assessed before and after the intervention with three static balance tests [bipedal stance (60″), Tandem Romberg stance (30″) and single leg stance (15″)] performed on a force platform, whereas the Mini-BESTest was also used to assess aspects of static and dynamic balance in the field.

Results

The IG significantly improved (p < 0.05) test scores of the Mini-BESTest and decreased the range and the root mean square of the centre of pressure displacement during the bipedal, Tandem Romberg and single leg stance, in contrast to the CG who showed no improvement.

Conclusion

The specific proprioceptive training programme improved the static balance of adults with ID and this could have a positive, significant impact in their daily life, as it may reduce the incidence of falls and relative injuries.

No abstract is available for this article.

ABSTRACT

Background

This study describes the proportion of Ontario adults with and without intellectual and developmental disabilities (IDD) who used community- and hospital-based healthcare in the first 2 years of the pandemic compared with the year pre–COVID-19.

Methods

Linked health administrative databases identified 87 341 adults with IDD and also adults without IDD living in Ontario, Canada. For each cohort, counts and proportions of adults who used different types of healthcare services were reported for the pre–COVID-19 year (16 March 2019 to 14 March 2020) and the first two COVID-19 years (15 March 2020 to 14 March 2021 and 15 March 2021 to 14 March 2022).

Results

Compared with the year prior to COVID-19, the proportion of adults with and without IDD who used health services was lower during the first COVID-19 year, but the likelihood of all types of visits increased during the second year. The likelihood of using homecare and of being hospitalized nearly returned to pre-pandemic levels. Virtual physician visits increased in each COVID-19 year from 5.2% prior to the pandemic to 13.0% in year 1 and 58.7% in year 2. For all years, the proportion of adults who used each service type was higher for those with IDD than without IDD.

Conclusions

For adults with and without IDD in Ontario, Canada, during the first two COVID-19 years healthcare use decreased for all service types, except for virtual physician visits. In the second year, healthcare use increased but did not reach pre–COVID-19 levels. In all years, adults with IDD were more likely to use services than other adults.

ABSTRACT

Introduction

Although existing research has explored both the benefits and risks associated with social internet use amongst people with intellectual disabilities (ID), a comprehensive understanding of the underlying reasons for this engagement is still lacking. This systematic review synthesizes literature investigating the reasons for social internet use amongst people with ID.

Methods

Eight electronic databases (Cinahl, Cochrane, Embase, ERIC, Google Scholar, Medline, PsycINFO and Web of Science) were systematically searched in June 2023 and November 2024 and screened using active machine learning techniques. Studies were considered for inclusion if they qualitatively described the reasons, motivations and personal opinions of people with ID regarding their social internet use in English and were published in peer-reviewed journals. Caregivers' insights were included if individuals could not verbally communicate directly. Only voluntary social internet use was considered; interventions were excluded unless preintervention views on social internet use were reported. Risk of bias was assessed using the Mixed Methods Appraisal Tool (MMAT; Hong et al. 2018). Data were extracted using the SPIDER tool and analysed using thematic synthesis.

Results

In total, 21 relevant articles were identified. Most studies described social internet use in Western contexts (n = 19), primarily amongst adults (n = 16). Four articles specifically addressed social internet use during COVID-19. Only seven studies explicitly reported participants' level of ID, with six focusing on mild-to-moderate ID and one on profound and multiple ID. Four themes emerged: a feeling of fitting in (n = 12), maintaining connections (n = 16), making new connections (n = 14) and enhancing autonomy and empowerment (n = 10).

Discussion

The findings underscore the importance of social internet use in fostering feelings of inclusion, connectedness and autonomy amongst people with ID. These insights can guide researchers and caregivers in developing tailored support strategies that both maximize the benefits and mitigate the risks of online social engagement for this population. By understanding the specific reasons behind social internet use, caregivers can offer more personalized guidance that aligns with the individual needs and preferences of people with ID. The review also highlights a need for future research to adhere to reporting guidelines to enhance transparency and quality in the field.

No abstract is available for this article.

ABSTRACT

Background

People with intellectual disabilities (IDs) require more vision care but encounter considerable challenges during eye examinations. Specialised clinics established specifically for people with IDs are generally limited. This study aims to evaluate primary family caregivers' willingness to pay (WTP) for specialised ophthalmology services designed for people with IDs.

Methods

Between 15 August and 5 October 2023, we conducted a face-to-face survey targeting primary family caregivers of people with IDs in two local authorities in Taiwan. We obtained a probability sample through stratified random sampling. A total of 657 family caregivers completed this survey, with a response rate of 82.6%. WTP was evaluated using a contingent valuation method in a hypothetical ophthalmology clinic conceptualised through a qualitative study. Two-part models were estimated.

Results

The WTP for specialised ophthalmology services designed for people with IDs varied from NT$96.9 (US$3.2) to NT$217.7 (US$7.3) for the lowest-income group (p < 0.001) and from NT$513.0 (US$17.1) to NT$648.6 (US$21.6) for the highest-income group (p < 0.001). Factors such as family income, self-reported financial satisfaction and concern for the ocular health of family members with IDs significantly influenced WTP.

Conclusions

Caregivers' WTP is low relative to the minimum operational costs of specialised ophthalmology clinics within the Taiwanese National Health Insurance scheme. This finding highlights the need for public funding to support such clinics and ensure that they can address the vision health disparity observed between people with and without IDs.