Journal of Intellectual Disability Research最新文献

{"title":"Beyond Uniform Impairment: Investigating Declarative Memory Profiles in Nonspecific Mild Intellectual Disability Using Latent Profile Analysis.","authors":"Urszula Sajewicz-Radtke, Bartosz M Radtke, Paweł Jurek, Michał Olech, Ariadna Łada-Maśko","doi":"10.1111/jir.70039","DOIUrl":"https://doi.org/10.1111/jir.70039","url":null,"abstract":"<p><strong>Background: </strong>Significant memory impairments are consistently observed in individuals with intellectual disabilities (ID), but considerable variability exists. This study investigated the heterogeneity of declarative memory in children and adolescents with nonspecific mild intellectual disability (NSID) to identify distinct memory profiles and potential predictors of this disability.</p><p><strong>Methods: </strong>A latent profile analysis (LPA) was conducted on a large sample (N = 999, including 114 with NSID) using six supplementary memory indices from the Test of Memory and Learning-Second Edition (TOMAL-2). A logistic regression analysis subsequently examined the predictive power of TOMAL-2 indices for NSID diagnosis.</p><p><strong>Results: </strong>LPA revealed two distinct memory profiles: a 'memory impaired group' (24% of the total sample) with below-average scores across all indices and a 'high-performers group' (76%) with consistently above-average scores. Individuals with NSID were significantly more likely to belong to the 'memory impaired group'. Logistic regression analysis revealed that lower scores on the Attention/Concentration Index, Sequential Memory Index and Verbal Delayed Recall Index were the strongest predictors of NSID. However, notably, 25% of individuals with mild NSID were classified in the 'high performers group', exhibiting typical or above-average memory scores across multiple indices.</p><p><strong>Conclusions: </strong>This study demonstrates significant heterogeneity in declarative memory among individuals with NSID, challenging the assumption of uniform impairment. The identified memory profiles and predictive indices offer valuable insights for more precise diagnostic assessment and the development of tailored interventions. Further research should investigate the factors contributing to this variability and explore the potential of these findings for improved support and educational strategies.</p><p><strong>Trial registration: </strong>NCT06215092.</p>","PeriodicalId":16163,"journal":{"name":"Journal of Intellectual Disability Research","volume":" ","pages":""},"PeriodicalIF":2.0,"publicationDate":"2025-09-03","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144992587","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":"Keynote Abstracts for Presentation at the SSBP 27th International Research Symposium and Educational Day, Amsterdam, the Netherlands","authors":"","doi":"10.1111/jir.70031","DOIUrl":"10.1111/jir.70031","url":null,"abstract":"","PeriodicalId":16163,"journal":{"name":"Journal of Intellectual Disability Research","volume":"69 9","pages":"749-752"},"PeriodicalIF":2.0,"publicationDate":"2025-09-02","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://onlinelibrary.wiley.com/doi/epdf/10.1111/jir.70031","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144957278","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":"Towards Personalised Care for Rare Genetic Disorders","authors":"Agnies M. van Eeghen,&nbsp;Erik Boot","doi":"10.1111/jir.70029","DOIUrl":"10.1111/jir.70029","url":null,"abstract":"","PeriodicalId":16163,"journal":{"name":"Journal of Intellectual Disability Research","volume":"69 9","pages":"729-731"},"PeriodicalIF":2.0,"publicationDate":"2025-09-02","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://onlinelibrary.wiley.com/doi/epdf/10.1111/jir.70029","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144957271","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":"Abstracts for Presentation at the SSBP 27th International Research Symposium and Educational Day, Amsterdam, the Netherlands","authors":"","doi":"10.1111/jir.70030","DOIUrl":"10.1111/jir.70030","url":null,"abstract":"","PeriodicalId":16163,"journal":{"name":"Journal of Intellectual Disability Research","volume":"69 9","pages":"732-748"},"PeriodicalIF":2.0,"publicationDate":"2025-09-02","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://onlinelibrary.wiley.com/doi/epdf/10.1111/jir.70030","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144957193","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":"Parental Stress and Family Quality of Life in Families of Individuals Living With Angelman Syndrome","authors":"Catherine Merton,&nbsp;Angela Gwaltney,&nbsp;Anna Booman,&nbsp;Sarah Nelson Potter,&nbsp;Anne C. Wheeler,&nbsp;Rene L. Barbieri-Welge,&nbsp;Lucia T. Horowitz,&nbsp;Rachel J. Hundley,&nbsp;Lynne M. Bird,&nbsp;Wen-Hann Tan,&nbsp;Anjali Sadhwani","doi":"10.1111/jir.70020","DOIUrl":"10.1111/jir.70020","url":null,"abstract":"<div>\u0000 \u0000 \u0000 <section>\u0000 \u0000 <h3> Background</h3>\u0000 \u0000 <p>Angelman syndrome (AS) is a developmental disorder caused by one of four molecular aetiologies. Affected individuals have intellectual disability (ID), limited speech, seizures and sleep problems. Parents of individuals with AS exhibit elevated stress compared to parents of individuals with other IDs. We examined parental stress and family quality of life (FQOL) over time in families of individuals living with AS.</p>\u0000 </section>\u0000 \u0000 <section>\u0000 \u0000 <h3> Methods</h3>\u0000 \u0000 <p>Data were collected in a natural history study of AS. The Parenting Stress Index, Third Edition (PSI) and the Beach Center FQOL Scale assessed parental stress and FQOL. Stress and FQOL were examined across AS molecular subtypes, and predictors were analysed using a generalised linear model. Relationships between parental stress and FQOL were examined using Pearson correlations and a stepwise mixed-linear model approach.</p>\u0000 </section>\u0000 \u0000 <section>\u0000 \u0000 <h3> Results</h3>\u0000 \u0000 <p>Our sample consisted of 231 families of individuals living with AS. Parental stress was clinically elevated and was highest in families of individuals with <i>UBE3A</i> pathogenic variants, whereas FQOL did not differ across subtypes in most domains. Increasing age predicted a decrease in parental stress but did not predict FQOL. Elevated parental stress was additionally predicted by maladaptive behaviours and child male sex, whereas lower FQOL was predicted by child male sex, parent marital status and family income. Parental stress had a small negative impact on FQOL.</p>\u0000 </section>\u0000 \u0000 <section>\u0000 \u0000 <h3> Conclusions</h3>\u0000 \u0000 <p>Stress is elevated in parents of individuals with AS across subtypes and has a negative impact on FQOL. Interventions to reduce stress have the potential to improve individual and family well-being.</p>\u0000 </section>\u0000 </div>","PeriodicalId":16163,"journal":{"name":"Journal of Intellectual Disability Research","volume":"69 9","pages":"822-839"},"PeriodicalIF":2.0,"publicationDate":"2025-09-02","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://onlinelibrary.wiley.com/doi/epdf/10.1111/jir.70020","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144957196","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":"Contrasting Relationships Between Anxiety and Intolerance of Uncertainty in Cornelia de Lange and Fragile X Syndromes.","authors":"Kayla Smith, Victoria Perry, Laura Groves, Joanna Moss, Chris Oliver, Eve Knight, Tom Patterson, Jacqui Rodgers, Jane Waite, Hayley Crawford","doi":"10.1111/jir.70024","DOIUrl":"https://doi.org/10.1111/jir.70024","url":null,"abstract":"<p><strong>Background: </strong>Cornelia de Lange syndrome (CdLS) and fragile X syndrome (FXS) are associated with co-occurring autism and anxiety. In autistic people, intolerance of uncertainty (IU) mediates the relationship between autistic characteristics and anxiety, but it is not known whether this relationship is evident in these genetic syndromes. Understanding the relationship between autism, anxiety and IU is essential to informing the theoretical frameworks of anxiety in rare genetic syndromes and improving clinical interventions.</p><p><strong>Method: </strong>Sixty participants with CdLS or FXS participated in a questionnaire-based study to examine the association between autistic characteristics, anxiety and IU.</p><p><strong>Results: </strong>IU mediated the association between autism and anxiety in participants with CdLS but not in participants with FXS.</p><p><strong>Conclusions: </strong>These results suggest that other factors may contribute to the autism-anxiety relationship in FXS, and highlight the merit of syndrome-specific approaches to the study of anxiety. Recommendations are made for intervention-based research to ameliorate anxiety in CdLS.</p>","PeriodicalId":16163,"journal":{"name":"Journal of Intellectual Disability Research","volume":" ","pages":""},"PeriodicalIF":2.0,"publicationDate":"2025-08-29","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144957236","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":"Healthcare Utilisation and Barriers and Facilitators of Healthcare Access for Young People With Intellectual Disability: A Systematic Review.","authors":"Felicia Kreps, Shalini Wijekulasuriya, Yvonne Zurynski, Rebecca Mitchell","doi":"10.1111/jir.70033","DOIUrl":"https://doi.org/10.1111/jir.70033","url":null,"abstract":"<p><strong>Background: </strong>Young people with intellectual disability may exhibit poorer general health, higher mortality rates and greater limitations from physical or mental illnesses compared to the general population. It is important to understand how this may relate to healthcare utilisation, including factors influencing healthcare access for young people with intellectual disability. This systematic review aimed to examine healthcare utilisation and to identify common barriers and facilitators of healthcare access for young people with intellectual disability.</p><p><strong>Method: </strong>Five databases (MEDLINE, Scopus, EMBASE, PsycINFO and CINAHL) were searched from 1 January 2013 to 31 October 2024 to identify articles that examined healthcare utilisation for young people (≤ 18 years old) with intellectual disability. Abstract and full-text screening were conducted by two reviewers; data from included articles were critically appraised.</p><p><strong>Results: </strong>Thirty-three studies were included for synthesis and focused on utilisation of hospital inpatient services (n = 22), emergency department (ED) services (n = 13), mental health or psychiatric services (n = 7) and mental health day programmes or outreach services (n = 1). Young people with intellectual disability generally had a higher proportion of hospital admissions, ED visits, hospital length of stay, mental health outpatient visits, mortality, 30-day hospital readmission and complications of care, compared to young people without intellectual disability. Common facilitators of healthcare access and engagement included having health insurance coverage and the use of visual aids and tools for the young person with intellectual disability. Barriers included low socio-economic status and poor provider communication and knowledge about intellectual disability.</p><p><strong>Conclusions: </strong>There are clear disparities in health service outcomes for young people with intellectual disability compared to the general population, which may be influenced by socio-cultural factors and access to knowledgeable and empathetic healthcare providers. Targeted education for healthcare providers may enhance the provision of high-quality care and improve healthcare utilisation and health outcomes for young people with intellectual disability.</p>","PeriodicalId":16163,"journal":{"name":"Journal of Intellectual Disability Research","volume":" ","pages":""},"PeriodicalIF":2.0,"publicationDate":"2025-08-25","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144957252","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":"Scale of Emotional Development-Questionnaire: A Systematic Approach to Improving Performance.","authors":"Mitchel Mesker, Jolanda Vonk, Suzanne D M Derks, Veerle Andries, Angelique van Lier-Weir, Paula S Sterkenburg","doi":"10.1111/jir.70019","DOIUrl":"https://doi.org/10.1111/jir.70019","url":null,"abstract":"<p><strong>Background: </strong>In people with intellectual disabilities, emotional development progresses more slowly or stagnates, which can result in challenging behaviours. The Scale of Emotional Development-Questionnaire was designed to chart people's own emotional development, providing insight into basic emotional needs and resilience, while reducing prejudice, enhancing self-awareness and improving emotional expression.</p><p><strong>Methods: </strong>The questionnaire was completed by 134 participants with moderate to borderline intellectual disabilities. Reliability, validity, internal structure and item performance were analysed to identify areas for improvement.</p><p><strong>Results: </strong>Preliminary analyses indicated the questionnaire captures key aspects of emotional development, with most items showing strong factor loadings (51.79%). However, multiple items may require refinement due to moderate loadings (30.00%), low loadings (18.21%), limited variance (3.93%) and negative-low correlations.</p><p><strong>Conclusion: </strong>The Scale of Emotional Development-Questionnaire is a promising self-report interview of emotional development, complementing the proxy perspective of the Scale of Emotional Development-Short. The findings highlight areas for improvement and the need for further research post-revision.</p>","PeriodicalId":16163,"journal":{"name":"Journal of Intellectual Disability Research","volume":" ","pages":""},"PeriodicalIF":2.0,"publicationDate":"2025-08-20","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144957201","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":"Dementia in Intellectual Disability: An Exploratory Investigation of Comorbidity Patterns and Diagnostic Outcomes.","authors":"Peer C Keller, Tanja Sappok","doi":"10.1111/jir.70016","DOIUrl":"https://doi.org/10.1111/jir.70016","url":null,"abstract":"<p><strong>Background: </strong>Dementia is more prevalent and tends to manifest earlier in individuals with intellectual disabilities (ID) compared to the general population. Acquiring specific knowledge about comorbidities and diagnostic findings in individuals with ID who have dementia, as opposed to those with ID without dementia, is essential. Such insights are crucial for enhancing the quality of care.</p><p><strong>Methods: </strong>The study was applied in a German outpatient clinic for people with ID and mental illnesses from February 2018 to September 2022. An exploratory comparison was conducted to identify differences in somatic and psychiatric comorbidities, laboratory results, cerebrospinal fluid results, neuroimaging, medication and challenging behaviour in people with ID with (n = 13, mean age: 54 years, 69% female) and without dementia (n = 73, mean age: 53 years, 48% female).</p><p><strong>Results: </strong>In this sample, persons with ID who have dementia are more likely to have Down syndrome and less likely to have affective disorders. They received antidementia drugs more often and atypical high-potency antipsychotics less often compared to persons with ID without dementia. All other clinical data showed no differences.</p><p><strong>Conclusions: </strong>Interestingly, no differences in somatic diseases (except Down syndrome) or laboratory and neuroimaging results could be found between people with ID with and without dementia. However, the diagnosis of dementia was associated with a reduced frequency of affective disorders and a reduced prescription of antipsychotics compared to the clinical sample without dementia. Due to the exploratory character of the study, replication in a much larger sample is necessary.</p>","PeriodicalId":16163,"journal":{"name":"Journal of Intellectual Disability Research","volume":" ","pages":""},"PeriodicalIF":2.0,"publicationDate":"2025-08-20","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144957209","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 Psychometric Properties of Emotional Development Assessment tools in Intellectual Disabilities: A Systematic Review.","authors":"Bethany Leal, Mark Hudson","doi":"10.1111/jir.70023","DOIUrl":"https://doi.org/10.1111/jir.70023","url":null,"abstract":"<p><strong>Background: </strong>People with intellectual disabilities can experience psychological distress and show behaviours of concern, such as self-injurious behaviour or physical aggression. One contributing factor is the degree to which their emotional needs are understood by those in their environment. This paper aims to review the psychometric properties of assessment tools measuring emotional development in individuals with intellectual disabilities.</p><p><strong>Methods: </strong>A systematic literature review was conducted, which included 5 databases and followed the PRISMA guidance (registration number: CRD42024553322). Seven assessment tools were included in this review: the SAED, SED-S, Brief SED-S, SED-R, and SED-R², SEO-Lukas and the Frankish model, and the psychometric properties were assessed in accordance with the COSMIN good measurement properties checklist.</p><p><strong>Results: </strong>Sixteen studies were included in this review. Internal consistency was assessed in six of the seven measures; validity was only assessed in the SAED and SED-S. Whilst both of these measures were considered reliable and valid, studies on the SAED had greater methodological quality, and the SED-S had a larger quantity of evidence.</p><p><strong>Conclusions: </strong>Both the SAED and the SED-S are psychometrically sound tools, based on the overall quality and sufficiency of the evidence. Further research should consider the usability, sensitivity and cross-cultural use, especially in UK populations.</p>","PeriodicalId":16163,"journal":{"name":"Journal of Intellectual Disability Research","volume":" ","pages":""},"PeriodicalIF":2.0,"publicationDate":"2025-08-12","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144835238","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}