Journal of Intellectual Disability Research最新文献

{"title":"Cholelithiasis prevalence and risk factors in individuals with severe or profound intellectual and motor disabilities","authors":"A. Watanabe,&nbsp;Y. Tomioka,&nbsp;Y. Okata,&nbsp;S. Yoshimura,&nbsp;S. Kumode,&nbsp;S. Iwabuchi,&nbsp;Y. Kameoka,&nbsp;Y. Takanarita,&nbsp;K. Uemura,&nbsp;Y. Samejima,&nbsp;Y. Kawasaki,&nbsp;Y. Bitoh","doi":"10.1111/jir.13113","DOIUrl":"10.1111/jir.13113","url":null,"abstract":"<div>\u0000 \u0000 \u0000 <section>\u0000 \u0000 <h3> Background</h3>\u0000 \u0000 <p>The prevalence and risk factors of cholelithiasis in individuals with severe or profound intellectual and motor disabilities (SPIMD) are poorly characterised. Thus, we aimed to investigate the prevalence and risk determinants of cholelithiasis in a cohort with SPIMD under medical care in a residential facility.</p>\u0000 </section>\u0000 \u0000 <section>\u0000 \u0000 <h3> Methods</h3>\u0000 \u0000 <p>We categorised 84 patients in a residential hospital for persons with SPIMD into groups: those with (Group CL) and without (Group N) cholelithiasis. Gallstones were detected via computed tomography, ultrasonography or both. We evaluated gastrostomy status, nutritional and respiratory support, constipation, and bladder and kidney stones. Data were significantly analysed using univariate and multivariate logistic regression analyses.</p>\u0000 </section>\u0000 \u0000 <section>\u0000 \u0000 <h3> Results</h3>\u0000 \u0000 <p>The prevalence rate of cholelithiasis in our SPIMD cohort was 27%. There were no significant differences in sex, age, weight, height, or Gross Motor Function Classification System between the two groups. However, more patients received enteral nutrition (39.13% vs. 6.56%; <i>P</i> = 0.000751) and were on ventilator support (56.52% vs. 19.67%; <i>P</i> = 0.00249) in Group CL than in Group N. Enteral nutrition [odds ratio (OR) 10.4, 95% confidence interval (CI) 1.98–54.7] and ventilator support (OR 20.0, 95% CI 1.99–201.0) were identified as independent risk factors for the prevalence of cholelithiasis in patients with SPIMD.</p>\u0000 </section>\u0000 \u0000 <section>\u0000 \u0000 <h3> Conclusions</h3>\u0000 \u0000 <p>Patients with SPIMD demonstrated an increased prevalence of cholelithiasis, with a notable association between nutritional tonic use and respiratory support. Therefore, to emphasise the need for proactive screening, it is crucial to devise diagnostic and therapeutic strategies specific to patients with SPIMD. Further investigation is essential to validate our findings and explore causative factors.</p>\u0000 </section>\u0000 </div>","PeriodicalId":16163,"journal":{"name":"Journal of Intellectual Disability Research","volume":"68 4","pages":"317-324"},"PeriodicalIF":3.6,"publicationDate":"2024-01-06","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"139111009","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":2,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}

{"title":"The relationship between the severity of intellectual and developmental disabilities (IDDs) in adults with IDDs and eating and drinking problems and nutritional status","authors":"M. E. Öztürk,&nbsp;N. Yabanci Ayhan","doi":"10.1111/jir.13114","DOIUrl":"10.1111/jir.13114","url":null,"abstract":"&lt;div&gt;\u0000 \u0000 \u0000 &lt;section&gt;\u0000 \u0000 &lt;h3&gt; Background&lt;/h3&gt;\u0000 \u0000 &lt;p&gt;Adults with intellectual and developmental disabilities (IDDs) experience eating, drinking and swallowing problems, such as chewing problems, choking, gagging, coughing during eating, aspiration and rumination syndrome, which may lead to poor nutritional status. This study aimed to determine the relationship between IDD levels, eating, drinking and swallowing problems and nutritional status in adults with IDDs.&lt;/p&gt;\u0000 &lt;/section&gt;\u0000 \u0000 &lt;section&gt;\u0000 \u0000 &lt;h3&gt; Methods&lt;/h3&gt;\u0000 \u0000 &lt;p&gt;The sample consisted of 71 participants (37 men and 34 women) with a mean age of 22.5 ± 7 years (range 18–60 years). Professionals classified intellectual disability as mild, moderate or severe. The Screening Tool of Feeding Problems scale was applied to the caregivers of adults with IDDs to identify eating, drinking and swallowing problems. Dietary intake was assessed using a 24-h dietary recall and a food and nutrition photograph catalogue. The researchers measured body weight, height and middle upper arm circumference. Body mass index was calculated. Four body mass index categories were determined: underweight (&lt;18.5 kg/m&lt;sup&gt;2&lt;/sup&gt;), normal weight (18.5–24.9 kg/m&lt;sup&gt;2&lt;/sup&gt;), overweight (25.0–29.9 kg/m&lt;sup&gt;2&lt;/sup&gt;) and obese (≥30 kg/m&lt;sup&gt;2&lt;/sup&gt;). Chi-squared tests were used to detect the relationship between IDD levels and eating and drinking problems, and analysis of variance tests were conducted to detect the relationship between IDD levels with anthropometric measurements and dietary intake.&lt;/p&gt;\u0000 &lt;/section&gt;\u0000 \u0000 &lt;section&gt;\u0000 \u0000 &lt;h3&gt; Results&lt;/h3&gt;\u0000 \u0000 &lt;p&gt;Participants had mild (42.3%; &lt;i&gt;n&lt;/i&gt; = 30), moderate (29.6%; &lt;i&gt;n&lt;/i&gt; = 21) or severe IDD (28.2%; &lt;i&gt;n&lt;/i&gt; = 20). They were underweight (12.7%; &lt;i&gt;n&lt;/i&gt; = 9), normal weight (59.2%; &lt;i&gt;n&lt;/i&gt; = 42) or overweight and/or obese (28.2%; &lt;i&gt;n&lt;/i&gt; = 20). Participants with severe IDD had significantly higher Screening Tool of Feeding Problems ‘nutrition-related behaviour’ and ‘eating and drinking skill deficit problem’ sub-scale scores than those with mild IDD. However, the groups had no significant difference in ‘food refusal and selectivity’ sub-scale scores. Participants with severe IDD also had anorexia prevalence similar to those with mild IDD. The groups did not significantly differ in anthropometric measurements, daily energy intake and macronutrient and micronutrient intake.&lt;/p&gt;\u0000 &lt;/section&gt;\u0000 \u0000 &lt;section&gt;\u0000 \u0000 &lt;h3&gt; Conclusions&lt;/h3&gt;\u0000 \u0000 &lt;p&gt;While adults with severe IDD had more eating and drinking skill deficits (e.g. chewing problems and independent eating difficulties) and nutrition-related behaviour problems than those with mild IDD, the eating, drinking and swallowing problems, which may critically affect","PeriodicalId":16163,"journal":{"name":"Journal of Intellectual Disability Research","volume":"68 4","pages":"325-339"},"PeriodicalIF":3.6,"publicationDate":"2024-01-06","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"139111011","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":2,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}

{"title":"Relationship between sensory processing patterns and gross motor function of children and adolescents with Down syndrome and typical development: a cross-sectional study","authors":"B. H. Brugnaro,&nbsp;M. F. Pauletti,&nbsp;C. R. G. Lima,&nbsp;B. N. Verdério,&nbsp;R. I. Fonseca-Angulo,&nbsp;B. Romão-Silva,&nbsp;A. C. de Campos,&nbsp;P. Rosenbaum,&nbsp;N. A. C. F. Rocha","doi":"10.1111/jir.13118","DOIUrl":"10.1111/jir.13118","url":null,"abstract":"<div>\u0000 \u0000 \u0000 <section>\u0000 \u0000 <h3> Background</h3>\u0000 \u0000 <p>Children and adolescents with Down syndrome (DS) may experience impairments in sensory and motor skills that can be interrelated. The purposes of this study were (i) to compare the sensory processing patterns and gross motor function between children and adolescents with DS and with typical development (TD) and (ii) to explore associations between these areas in both DS and TD groups.</p>\u0000 </section>\u0000 \u0000 <section>\u0000 \u0000 <h3> Method</h3>\u0000 \u0000 <p>This cross-sectional study involved a sample size of 25 participants with DS (mean age 10.24 ± 2.04 years) and 25 participants with TD (mean age 10.04 ± 2.82 years). The sensory processing patterns were assessed using the Sensory Profile Second Version questionnaire, and the gross motor function was measured with the Gross Motor Function Measure (GMFM-88) dimensions (D) <i>standing</i>, and (E) <i>walking, running, and jumping</i>. Differences between groups were tested using the Mann–Whitney test, and the relationship between the variables was examined using Spearman's correlation tests, with a significance level set at 5%.</p>\u0000 </section>\u0000 \u0000 <section>\u0000 \u0000 <h3> Results</h3>\u0000 \u0000 <p>Children with DS showed greater difficulties with sensory processing than TD children in Seeker (<i>P</i> &lt; 0.001), Avoider (<i>P</i> &lt; 0.001), Sensitivity (<i>P</i> &lt; 0.001), Registration (<i>P</i> &lt; 0.001), Auditory (<i>P</i> &lt; 0.001), Touch (<i>P</i> = 0.001), Movements (<i>P</i> = 0.001), Oral (<i>P</i> = 0.028), Conduct (<i>P</i> = 0.005), Socioemotional (<i>P</i> &lt; 0.001), and Attentional (<i>P</i> &lt; 0.001) domains. Additionally, children with DS presented lower gross motor function than TD in GMFM-88, <i>standing</i> (<i>P</i> &lt; 0.001) and <i>walking, running, and jumping</i> (<i>P</i> &lt; 0.001). Correlations were found between greater difficulties with sensory processing in Touch and lower gross motor function in <i>walking, running, and jumping</i> for the DS group.</p>\u0000 </section>\u0000 \u0000 <section>\u0000 \u0000 <h3> Conclusion</h3>\u0000 \u0000 <p>Our results suggest there are more difficulties in sensory processing patterns and gross motor function in children with DS than in TD. Also, there is a single association between more difficulties in sensory processing and less well-developed motor function in the DS group. Therefore, a comprehensive assessment of all these aspects should be performed in children and adolescents with DS, along with the provision of relevant interventions addressing specific needs.</p>\u0000 </section>\u0000 </div>","PeriodicalId":16163,"journal":{"name":"Journal of Intellectual Disability Research","volume":"68 4","pages":"358-368"},"PeriodicalIF":3.6,"publicationDate":"2024-01-06","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"139111010","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":2,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}

{"title":"A profile of spatial abilities in people with Down syndrome","authors":"A. Bracken,&nbsp;J. Hauss,&nbsp;S. Grinshpun,&nbsp;D. Lasc,&nbsp;A. Hershkovich,&nbsp;Y. Yang","doi":"10.1111/jir.13111","DOIUrl":"10.1111/jir.13111","url":null,"abstract":"<div>\u0000 \u0000 \u0000 <section>\u0000 \u0000 <h3> Background</h3>\u0000 \u0000 <p>Spatial abilities are fundamental cognitive abilities, have direct applications in daily life, serve as a cognitive foundation for many other complex skills and are used in many specialty jobs. The current study aimed to systematically and comprehensively evaluate the spatial abilities of individuals with Down syndrome (DS) relative to mental ability-matched typically developing (TD) children based on Newcombe and Shipley's double-dimension theoretical framework for classifying spatial abilities.</p>\u0000 </section>\u0000 \u0000 <section>\u0000 \u0000 <h3> Methods</h3>\u0000 \u0000 <p>Forty adolescents and young adults with DS and 40 TD children completed a nonverbal intelligence test (Raven's), two measures of static–extrinsic skills (water-level task and cart task), two measures of static–intrinsic skills (figure ground and form completion), two measures of dynamic–extrinsic skills (three mountains task and dog task) and two measures of dynamic–intrinsic spatial skills (mental rotation task and block design task).</p>\u0000 </section>\u0000 \u0000 <section>\u0000 \u0000 <h3> Results</h3>\u0000 \u0000 <p>Participants with DS showed reduced performance on two dynamic–intrinsic tasks and one static–extrinsic task (i.e. cart task) relative to TD children. Performances were similar in two dynamic–extrinsic tasks and two static–intrinsic tasks. Analyses of composite accuracy for each spatial category further confirmed deficits in dynamic–intrinsic and static–extrinsic categories for people with DS relative to TD children.</p>\u0000 </section>\u0000 \u0000 <section>\u0000 \u0000 <h3> Conclusions</h3>\u0000 \u0000 <p>Our results showed an uneven profile of spatial abilities in people with DS relative to ability-matched TD children with particular weaknesses in comprehending and manipulating dynamic–intrinsic and static–extrinsic spatial relations. Furthermore, our research has important clinical implications for more targeted interventions to improve spatial abilities in people with DS.</p>\u0000 </section>\u0000 </div>","PeriodicalId":16163,"journal":{"name":"Journal of Intellectual Disability Research","volume":"68 3","pages":"223-236"},"PeriodicalIF":3.6,"publicationDate":"2023-12-10","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"138584765","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":2,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}

{"title":"Cancer risk-factor and symptom awareness among adults with intellectual disabilities, paid and unpaid carers, and healthcare practitioners: a scoping review","authors":"N. Gil,&nbsp;A. Cox,&nbsp;K. L. Whitaker,&nbsp;R. S. Kerrison","doi":"10.1111/jir.13110","DOIUrl":"10.1111/jir.13110","url":null,"abstract":"<div>\u0000 \u0000 \u0000 <section>\u0000 \u0000 <h3> Background</h3>\u0000 \u0000 <p>The physical health of people with intellectual disabilities (ID) has been identified as an area of ongoing concern and priority. Research has increasingly focused on cancer, with studies indicating that people with ID are at an increased risk of cancer and of mortality, compared with the general population. This review aims to systematically identify and synthesise the published academic literature exploring cancer risk-factor and symptom awareness among people with IDs, carers and healthcare professionals.</p>\u0000 </section>\u0000 \u0000 <section>\u0000 \u0000 <h3> Methods</h3>\u0000 \u0000 <p>In line with Arksey and O'Malley's (2005) framework for scoping reviews, five incremental stages were followed: (1) identifying research question, (2) identifying relevant studies, (3) study selection, (4) extracting and charting of data, and (5) collating, summarising and reporting results. Findings were reported according to the Preferred Reporting Items for Systematic Reviews and Meta-analyses extension for scoping reviews (PRISMA-Scr).</p>\u0000 </section>\u0000 \u0000 <section>\u0000 \u0000 <h3> Results</h3>\u0000 \u0000 <p>The search strategy identified 352 records, 16 records met all eligibility criteria and were included for review. The studies address a range of areas including knowledge and awareness of cancer risk-factors and symptoms and interventions to promote awareness of cancer.</p>\u0000 </section>\u0000 \u0000 <section>\u0000 \u0000 <h3> Conclusions</h3>\u0000 \u0000 <p>Cancer risk-factor and symptom awareness is low among adults with ID, paid and unpaid carers and healthcare practitioners (HCPs). Theoretically underpinned, co-designed tools and interventions to improve awareness are lacking. There is uncertainty surrounding how to best support people with ID in raising cancer awareness, even within the professional healthcare environment. There is a predominance of research on breast cancer awareness. Future studies focusing on other cancers are needed to build a complete picture of awareness among adults with IDs, paid and unpaid carers, and HCPs.</p>\u0000 </section>\u0000 </div>","PeriodicalId":16163,"journal":{"name":"Journal of Intellectual Disability Research","volume":"68 3","pages":"193-211"},"PeriodicalIF":3.6,"publicationDate":"2023-12-06","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://onlinelibrary.wiley.com/doi/epdf/10.1111/jir.13110","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"138498614","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":2,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}

{"title":"Challenging behaviour and its correlates in preschool-aged children with an intellectual disability in Saudi Arabia","authors":"S. Alarifi,&nbsp;L. Denne,&nbsp;R. P. Hastings","doi":"10.1111/jir.13109","DOIUrl":"10.1111/jir.13109","url":null,"abstract":"<div>\u0000 \u0000 \u0000 <section>\u0000 \u0000 <h3> Background</h3>\u0000 \u0000 <p>Young children with an intellectual disability have a higher risk of developing challenging behaviour (CB). Early identification of risk factors for CB allows for earlier intervention. The aim of the current study was to assess the prevalence and correlates of CB in preschool-aged children with an intellectual disability in Riyadh (Saudi Arabia).</p>\u0000 </section>\u0000 \u0000 <section>\u0000 \u0000 <h3> Methods</h3>\u0000 \u0000 <p>One hundred twenty parents of preschool-aged (3–6 years old) children who had been diagnosed (DSM-5 criteria) with an intellectual disability completed an online cross-sectional survey that included demographic, CB and child adaptive skills measures. The relationship between CB and 15 potential correlates (e.g. gender and degree of disability) was examined using independent samples t-tests and chi-squared tests.</p>\u0000 </section>\u0000 \u0000 <section>\u0000 \u0000 <h3> Results</h3>\u0000 \u0000 <p>Most preschool-aged (3–6 years old) children with an intellectual disability exhibited CB (78.8%, 95% CI [70.3, 85.8]), with a 63.2% prevalence rate for self-injurious behaviours (95% C [53.8, 72.0]), a 57.6% rate for aggressive destructive behaviours (95% CI [48.2, 66.7]) and a 25% rate for stereotypy (95% CI [17.7, 34.0]). The likelihood of a child engaging in self-injurious and stereotyped behaviours was higher in those with autism and intellectual disability. Children with Down syndrome displayed fewer stereotyped behaviours. Low adaptive skill levels were associated with increased overall CB, self-injurious and stereotyped behaviours.</p>\u0000 </section>\u0000 \u0000 <section>\u0000 \u0000 <h3> Conclusions</h3>\u0000 \u0000 <p>The identified correlates of CB in this population and cultural context align with the international evidence base. Findings have implications for the importance of early systematic screening of CB in preschool-aged children in Saudi Arabia and other similar contexts. Preventative measures are suggested for preschool-aged children with an intellectual disability who are more likely to demonstrate CB, such as those with autism and poor adaptive behaviours.</p>\u0000 </section>\u0000 </div>","PeriodicalId":16163,"journal":{"name":"Journal of Intellectual Disability Research","volume":"68 3","pages":"264-276"},"PeriodicalIF":3.6,"publicationDate":"2023-12-05","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://onlinelibrary.wiley.com/doi/epdf/10.1111/jir.13109","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"138487761","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":2,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}

{"title":"Clinical characteristics of people with intellectual disability admitted to hospital with constipation: identifying possible specific high-risk factors","authors":"R. Laugharne,&nbsp;M. Wilcock,&nbsp;J. Rees,&nbsp;D. Wainwright,&nbsp;N. Newton,&nbsp;J. Sterritt,&nbsp;S. Badger,&nbsp;R. Bishop,&nbsp;P. Bassett,&nbsp;R. Shankar","doi":"10.1111/jir.13108","DOIUrl":"10.1111/jir.13108","url":null,"abstract":"<div>\u0000 \u0000 \u0000 <section>\u0000 \u0000 <h3> Background</h3>\u0000 \u0000 <p>People with intellectual disabilities (ID) die on an average 20 years earlier to the general population. They have higher rates of multimorbidity and polypharmacy. Around 25% of people with ID report chronic constipation. The England Learning Disabilities Mortality Review found that nearly 25% of deaths identified constipation as a long-term health problem. However, the likely risk factors for constipation related harm are poorly enumerated. We sought to identify possible specific high-risk factors by examining the clinical characteristics of people with ID admitted to hospital with constipation.</p>\u0000 </section>\u0000 \u0000 <section>\u0000 \u0000 <h3> Methods</h3>\u0000 \u0000 <p>Data of people with ID admitted with constipation in two general hospitals covering a population of 1.3 million from 2017 to 2022 were reported using the STROBE guideline for cohort studies. Collected data included age, gender, intellectual disability severity, recorded medication, presenting complaint and co-morbidities. The medication anticholinergic burden was calculated using the anticholinergic burden scale. Continuous variables were summarised by mean and standard deviation if normally distributed, with categorical variables summarised by the number and percentage in each category.</p>\u0000 </section>\u0000 \u0000 <section>\u0000 \u0000 <h3> Results</h3>\u0000 \u0000 <p>Of 46 admissions (males 52%), 57% had moderate to profound ID, 37% had epilepsy, 41% prescribed antiseizure medication (ASM) and 45% were on laxatives. Average age was 46 years. The anticholinergic burden score mean was 2.3 and median, one.</p>\u0000 </section>\u0000 \u0000 <section>\u0000 \u0000 <h3> Conclusions</h3>\u0000 \u0000 <p>We can hypothesise that people with more severe ID, suffering from epilepsy and on ASM may be more at risk of developing severe constipation. Some admissions may be avoided with earlier use of laxatives in the community.</p>\u0000 </section>\u0000 </div>","PeriodicalId":16163,"journal":{"name":"Journal of Intellectual Disability Research","volume":"68 3","pages":"277-284"},"PeriodicalIF":3.6,"publicationDate":"2023-11-30","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://onlinelibrary.wiley.com/doi/epdf/10.1111/jir.13108","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"138460370","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":2,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}

{"title":"Child characteristics associated with child quality of life and parenting stress in Angelman syndrome","authors":"D. A. Hagenaar,&nbsp;K. G. C. B. Bindels-de Heus,&nbsp;K. Lubbers,&nbsp;L. W. ten Hoopen,&nbsp;A. B. Rietman,&nbsp;P. F. A. de Nijs,&nbsp;M. H. J. Hillegers,&nbsp;H. A. Moll,&nbsp;M. C. Y. de Wit,&nbsp;G. C. Dieleman,&nbsp;S. E. Mous","doi":"10.1111/jir.13106","DOIUrl":"10.1111/jir.13106","url":null,"abstract":"<div>\u0000 \u0000 \u0000 <section>\u0000 \u0000 <h3> Background</h3>\u0000 \u0000 <p>Angelman syndrome (AS) is a rare neurodevelopmental disorder characterised by severe intellectual disability, movement disorder, epilepsy, sleeping problems, and behavioural issues. Little is known on child health-related quality of life (HRQoL) in AS. AS family studies have reported elevated parenting stress and a high impact of the child's syndrome on the parent. It is unclear which factors influence child HRQoL and parenting stress/impact in AS.</p>\u0000 </section>\u0000 \u0000 <section>\u0000 \u0000 <h3> Methods</h3>\u0000 \u0000 <p>We collected data prospectively through standardised clinical assessments of children with AS at the ENCORE Expertise centre for Angelman Syndrome at the Erasmus MC Sophia Children's Hospital. A linear regression analysis was conducted for the following outcome variables: (1) child HRQoL (Infant and Toddler Quality of Life Questionnaire); (2) the impact of the child's syndrome on the parent (Infant and Toddler Quality of Life Questionnaire); and (3) parenting stress (Parenting Stress Index). Predictor variables were child genotype, epilepsy, sleeping problems (Sleep Disturbance Scale for Children), cognitive developmental level (Bayley Cognition Scale), autistic features (Autism Diagnostic Observation Schedule) and emotional/behavioural problems (Child Behaviour Checklist). Covariates were sex, age and socio-economic status.</p>\u0000 </section>\u0000 \u0000 <section>\u0000 \u0000 <h3> Results</h3>\u0000 \u0000 <p>The study sample consisted of 73 children with AS, mean age = 9.1 years, range = 2–18 years. Emotional/behavioural problems were the strongest significant predictor of lowered child HRQoL. Internalising problems were driving this effect. In addition, having the deletion genotype and higher age was related to lower child HRQoL. Sleeping problems were related to a higher impact of the child's syndrome on the parent. Finally, emotional/behavioural problems were associated with higher parenting stress. Cognitive developmental level, autistic features and epilepsy were not a significant predictor of child HRQoL and parenting stress/impact.</p>\u0000 </section>\u0000 \u0000 <section>\u0000 \u0000 <h3> Conclusions</h3>\u0000 \u0000 <p>These results suggest that interventions aimed at increasing child HRQoL and decreasing parenting stress/impact in AS should focus on child emotional/behavioural problems and sleeping problems, using a family-centred approach.</p>\u0000 </section>\u0000 </div>","PeriodicalId":16163,"journal":{"name":"Journal of Intellectual Disability Research","volume":"68 3","pages":"248-263"},"PeriodicalIF":3.6,"publicationDate":"2023-11-27","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://onlinelibrary.wiley.com/doi/epdf/10.1111/jir.13106","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"138444905","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":2,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}

{"title":"The physical and psychosocial impact of a school-based running programme for adolescents with disabilities","authors":"J. Campagna,&nbsp;K. Czyszczon,&nbsp;J. Little,&nbsp;C. Selby,&nbsp;L. Wick,&nbsp;D. Ferreira,&nbsp;K. Oriel","doi":"10.1111/jir.13104","DOIUrl":"10.1111/jir.13104","url":null,"abstract":"<div>\u0000 \u0000 \u0000 <section>\u0000 \u0000 <h3> Background</h3>\u0000 \u0000 <p>Adolescents with disabilities have fewer opportunities to participate in community-based fitness programmes. The purpose of this study was to examine the impact of a school-based running programme at a local middle school in Lebanon, Pennsylvania, on fitness and quality of life (QoL) in children with physical and cognitive disabilities in a life-skills classroom.</p>\u0000 </section>\u0000 \u0000 <section>\u0000 \u0000 <h3> Methods</h3>\u0000 \u0000 <p>Nineteen adolescents with diagnosed disabilities including intellectual disability (ID), autism spectrum disorder and Down syndrome were recruited from three life-skills classrooms to participate in a school-based running programme. The programme was designed to be implemented two times/week for 6 weeks by classroom teachers/aides. Physical therapy faculty and students developed the programme and assisted with implementation. Each session lasted 30 min, consisting of a warm-up and cooldown, relay races, games and timed runs. Pre- and post-test measures included physiological cost index (PCI) and Paediatric Quality of Life Inventory™ (PedsQL™). Pre- and post-test data were compared using Wilcoxon signed rank tests. Each week participants also completed a training log to reflect on the activity for the day.</p>\u0000 </section>\u0000 \u0000 <section>\u0000 \u0000 <h3> Results</h3>\u0000 \u0000 <p>Participants demonstrated significant improvements in PCI (<i>P</i> = 0.028) and the PedsQL™ (<i>P</i> = 0.008) following the running programme.</p>\u0000 </section>\u0000 \u0000 <section>\u0000 \u0000 <h3> Conclusions</h3>\u0000 \u0000 <p>Results of this study suggest that participation in a 6-week school-based running programme may improve fitness and QoL in adolescents with disabilities.</p>\u0000 </section>\u0000 </div>","PeriodicalId":16163,"journal":{"name":"Journal of Intellectual Disability Research","volume":"68 2","pages":"181-192"},"PeriodicalIF":3.6,"publicationDate":"2023-11-20","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"138176325","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":2,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}

{"title":"Functional fitness of adults with Down syndrome: a longitudinal study","authors":"P. H. Boer","doi":"10.1111/jir.13107","DOIUrl":"10.1111/jir.13107","url":null,"abstract":"<div>\u0000 \u0000 \u0000 <section>\u0000 \u0000 <h3> Background</h3>\u0000 \u0000 <p>Individuals with Down syndrome (DS) are born with and develop many health-related complications. The purpose of this study was to determine the longitudinal functional fitness profile of adults with DS.</p>\u0000 </section>\u0000 \u0000 <section>\u0000 \u0000 <h3> Methods</h3>\u0000 \u0000 <p>The functional fitness of adults with DS was tested twice, 12 years apart. Sixty-six adults with DS were tested for body mass, stature and 10 functional fitness tests. Data were categorised according to gender and age-specific categories.</p>\u0000 </section>\u0000 \u0000 <section>\u0000 \u0000 <h3> Results</h3>\u0000 \u0000 <p>Static balance, shoulder flexibility, trunk strength and aerobic capacity deteriorated significantly with medium to large effect sizes for both DS men and women (most age categories). For women, dynamic balance deteriorated significantly, and for men, leg- and upper body-strength deteriorated significantly.</p>\u0000 </section>\u0000 \u0000 <section>\u0000 \u0000 <h3> Conclusions</h3>\u0000 \u0000 <p>Practitioners working in the field of adapted physical activity should take cognisance of the functional fitness ageing profile of adults with DS and timeously develop habitual physical activity interventions to reduce the effect of accelerated ageing experienced by this population.</p>\u0000 </section>\u0000 </div>","PeriodicalId":16163,"journal":{"name":"Journal of Intellectual Disability Research","volume":"68 3","pages":"237-247"},"PeriodicalIF":3.6,"publicationDate":"2023-11-15","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://onlinelibrary.wiley.com/doi/epdf/10.1111/jir.13107","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"107591464","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":2,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}