Journal of Intellectual Disability Research最新文献

{"title":"Specialist Psychiatric Bed Utilisation by People With Intellectual Disabilities and Autistic People: A Time-Series Analysis Using the English Assuring Transformation Dataset.","authors":"Atiyya Nisar, Paul A Thompson, Harm Boer, Haider Al-Delfi, Peter E Langdon","doi":"10.1111/jir.70001","DOIUrl":"https://doi.org/10.1111/jir.70001","url":null,"abstract":"<p><strong>Background: </strong>Using nationally available anonymised and aggregated English data, we examined specialist and nonspecialist psychiatric bed utilisation by people with intellectual disabilities and/or autism.</p><p><strong>Methods: </strong>Using data about specialist psychiatric bed utilisation from the Assuring Transformation Dataset, from March 2015 to January 2024, we applied linear regression (with moving average or autoregressive errors) to explore the relationships between a set of outcome variables (e.g., number of inpatients and length of stay) and a set of sociodemographic, clinical and service-related predictor variables (e.g., age, ethnicity, admission source, legal status, admission source, discharge destination, Care (Education) and Treatment Reviews) over time. Comparisons were made with data from the Mental Health Services Data Set about nonspecialist psychiatric bed utilisation.</p><p><strong>Results: </strong>Over time, there was an average reduction of 8.07 inpatients per month. This reduction was due to a reduction in the number with a length of stay longer than 2 years, and fewer inpatients with intellectual disabilities without autism over time, rather than fewer autistic inpatients without intellectual disabilities; instead, the number of autistic inpatients increased by 6.02 per month. However, overall, there were fewer inpatients in specialist psychiatric beds than in nonspecialist beds by an average of 877 patients, and the number in specialist beds reduced faster than the number in nonspecialist beds over time. We found that more hospital spells were associated with more inpatients older than 18, more detentions under Part III of the Mental Health Act, more inpatients not known to the local authority, and an increased number of White inpatients. More admissions were associated with fewer discharges, while those with a hospital stay longer than 2 years were less likely to have had a postadmission Care (Education) and Treatment Reviews and were more likely to use advocacy.</p><p><strong>Conclusions: </strong>The number of inpatients with intellectual disabilities in specialist psychiatric beds continues to decline over time, while the number of autistic inpatients without intellectual disabilities is increasing. Future research should utilise participant-level data to explore patient long-term trajectories.</p>","PeriodicalId":16163,"journal":{"name":"Journal of Intellectual Disability Research","volume":" ","pages":""},"PeriodicalIF":2.1,"publicationDate":"2025-06-12","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144284939","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":"Acoustics-Derived Home-Based Sleep Study for the Diagnosis of Obstructive Sleep Apnoea in Adults With Intellectual Disabilities: A Feasibility Study.","authors":"Lachlan Stranks, Mohammad Abdulsalam, David O'Regan, Joerg Steier, Alanna Hare","doi":"10.1111/jir.70000","DOIUrl":"https://doi.org/10.1111/jir.70000","url":null,"abstract":"<p><strong>Background: </strong>Adults with intellectual disabilities (IDs) face significant health inequalities and reduced life expectancy compared to the general population. Sleep disorders, including obstructive sleep apnoea (OSA), are highly prevalent in this population; however, standard diagnostic modalities may be poorly tolerated and many people may remain undiagnosed. The aim of this study was to assess the feasibility of using an acoustic-derived home-based sleep study to diagnose OSA in adults with ID who were unable to tolerate standard inpatient or home sleep studies.</p><p><strong>Method: </strong>This was a prospective feasibility study (GSTT/2024/15803). Participants were identified from two tertiary outpatient sleep departments in London, UK. Eligible participants were aged ≥ 17 years with a diagnosed ID. Inpatient or existing home studies had not been tolerated or had been declined. Participants wore a small diagnostic sensor, the AcuPebble SA100 (Acurable Ltd., London, UK) for one to two nights in their own home. Baseline symptom screening and quality of life questionnaires were administered. Semiqualitative and qualitative feedback was obtained from participants. The primary outcome was successful completion of testing, defined as ≥ 240 min of recording on at least one night. Quantitative diagnostic parameters were reviewed in a multidisciplinary team (MDT) meeting to formulate a management plan. Data are presented as mean (standard deviation), unless otherwise specified.</p><p><strong>Results: </strong>Seventeen people were screened, and 10 agreed to participate in the study (3F/7M). Eight participants (80%) successfully completed testing (2F/6M, 4 severe ID, age 29.7 [15.4] years, body mass index, BMI 27.1 [5.9] kg/m², apnoea hypopnoea index, median AHI 5.5 [IQR 7.8] ev/h). Of these, five participants (63%) were found to have OSA. Feedback was favourable with an average total satisfaction score of 14.0 (4.8) out of 20 points.</p><p><strong>Conclusion: </strong>In a cohort of participants with ID who were unable to tolerate standard sleep diagnostics to assess OSA, an acoustic-derived home-based sleep study may provide a solution to achieve clinically diagnostic recordings.</p>","PeriodicalId":16163,"journal":{"name":"Journal of Intellectual Disability Research","volume":" ","pages":""},"PeriodicalIF":2.1,"publicationDate":"2025-06-11","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144266434","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":"Lifting Limits: The Impact of Strength Training in Down Syndrome-A Systematic Review and Meta-Analysis.","authors":"Luis Iglesias-Díaz, Susana López-Ortiz, Celia García-Chico, Alejandro Santos-Lozano, Javier González-Lázaro","doi":"10.1111/jir.13259","DOIUrl":"https://doi.org/10.1111/jir.13259","url":null,"abstract":"<p><strong>Background: </strong>People with Down syndrome (DS) may exhibit several musculoskeletal disorders, including alterations in muscle tone and activation. Strength training could mitigate the loss of muscle strength and, therefore, improve strength values in this population. Additionally, it may influence health-related outcomes such as physical function, body composition and biochemical markers.</p><p><strong>Objective: </strong>This systematic review and meta-analysis of randomised controlled trials (RCTs) aimed to analyse the characteristics and effects of strength training in people with DS.</p><p><strong>Methods: </strong>A search was conducted from inception to 22 April 2025. The methodological quality of the included RCTs was assessed using the 15-item Tool for the assEssment of Study qualiTy and reporting in EXercise (TESTEX). In addition, the risk of bias was assessed using the Cochrane's risk of bias 2 (RoB2).</p><p><strong>Results: </strong>A total of 10 RCTs (n = 233 participants) were included in the systematic review, of which three (n = 111 participants) could be meta-analysed. The pooled effect showed statistically significant benefits for upper (mean difference [MD] = 5.66 kg, 95% CI 2.42-8.91) and lower (MD = 20.43 kg, 95% CI 1.76-39.10) body strength. The TESTEX scores for most RCTs ranged from 3 to 12 points. The risk of bias analysis indicated that eight RCTs had a low risk of bias, whereas the remaining studies were classified as high risk.</p><p><strong>Conclusion: </strong>Strength training may significantly improve muscle strength in people with DS. However, further research is needed to assess the long-term effects on physical function, body composition and biochemical markers.</p>","PeriodicalId":16163,"journal":{"name":"Journal of Intellectual Disability Research","volume":" ","pages":""},"PeriodicalIF":2.1,"publicationDate":"2025-06-09","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144258214","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":"Co-Occurring Medical Conditions in Over 2300 Children With Down Syndrome at a Down Syndrome Multispecialty Clinic.","authors":"Francis Hickey, Liz Maastricht, Kristine Wolter-Warmerdam, Dee Daniels, Bailey Herfindahl, Karen Kelminson","doi":"10.1111/jir.13257","DOIUrl":"https://doi.org/10.1111/jir.13257","url":null,"abstract":"<p><strong>Background: </strong>Children with Down syndrome (DS) have an increased frequency of co-occurring medical complications compared to the typically developing population; however, incidence rates of co-occurring medical conditions in a large paediatric population-based study are limited. The goal of this study was to further investigate the frequencies of concomitant medical conditions, as well as newborn complications in children with DS.</p><p><strong>Methods: </strong>This is a retrospective, large cohort review of children with DS (n = 2321) receiving care at a referral centre specialising in DS to identify common patterns of co-occurring conditions in children and young adults with DS. Participants from birth to 24 years of age received care from the Anna and John J. Sie Center for Down Syndrome at Children's Hopsital Colorado, which serves 88% of the State of Colorado's paediatric population with DS. Records reviewed included demographics, birth history, and medical conditions of each patient.</p><p><strong>Results: </strong>Neonatal complications requiring admission to the neonatal intensive care unit (NICU) were present in 80.0% of the population. Complications leading to these admissions include oxygen requirement (53.7%), feeding problems (49.7%), respiratory distress syndrome (14.7%), and persistent pulmonary hypertension of the newborn (12.6%). Incidence of medical complications for children and young adults with DS includes obstructive sleep apnea (56.8%), cardiac defect requiring surgical repair (48.9%), feeding problems (42.3%), and dysphagia (26.2%). Recently identified co-occurring conditions lacking in the literature, such as keratoconus (3.6%) and Wolff-Parkinson-White syndrome (3.4%), are also reported. Testing, labs and procedures were also common amongst children to meet American Academy of Pediatrics (AAP) DS Guidelines.</p><p><strong>Conclusion: </strong>Children with DS have unique phenotypic clinical patterns of co-occurring conditions and medical complications. Some conditions present clinically in the neonatal period and require close monitoring and anticipation for a higher level of care for newborns with DS. Our results, using a large sample of over 2300 patients, provide information to improve evidence-based early identification and timely intervention for children and young adults with DS.</p>","PeriodicalId":16163,"journal":{"name":"Journal of Intellectual Disability Research","volume":" ","pages":""},"PeriodicalIF":2.1,"publicationDate":"2025-06-04","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144216082","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":"Reliability of the Dutch Version of the Matson Evaluation of Drug Side Effects in People With Intellectual Disabilities.","authors":"Maria Louise Dieudonnée Hoekstra-van Duijn, Maureen Bernardina Gerardus Wissing, Patrick Jan Eugène Bindels, Dederieke Anne Maria Maes-Festen","doi":"10.1111/jir.13256","DOIUrl":"https://doi.org/10.1111/jir.13256","url":null,"abstract":"<p><strong>Background: </strong>People with intellectual disabilities often use psychotropic medications. Recognising side effects in this population can be challenging while they can negatively impact the quality of life. An accurate screening instrument is crucial to identify side effects of psychotropic medication in people with intellectual disabilities. For this purpose, the Matson Evaluation of Drug Side Effects (MEDS) is the most reliable and well-researched instrument. We translated the MEDS into Dutch, considering that the Dutch version of the MEDS must be accurate and aligned with its intended meaning, avoiding multiple interpretations; this study aimed to assess its intrarater and interrater reliabilities in people with intellectual disabilities using psychotropic medication.</p><p><strong>Methods: </strong>A certified medical translation agency performed translation and back-translation of the MEDS. Two researchers administered the MEDS three times on the same day. Participants were people with intellectual disabilities using psychotropic medication. Demographic and medical data were collected via questionnaires. Scoring and calculation of total domain scores, severity domain scores, duration domain scores and composite scale scores followed the MEDS manual. Intraclass correlation coefficients (ICC) and 95% confidence intervals of these scores were used to assess intrarater and interrater reliabilities.</p><p><strong>Results: </strong>The study sample included 40 adults with intellectual disabilities. Intrarater reliability was good to excellent for total domain scores (ICCs ranging from 0.873 to 1.000) and excellent for the total composite scale score (ICC = 0.945). Interrater reliability was moderate to excellent for total domain scores (ICCs ranging from 0.713 to 0.922) and good for the total composite scale score (ICC = 0.894).</p><p><strong>Conclusions: </strong>This study demonstrated moderate to excellent intrarater and interrater reliabilities for the Dutch version of the MEDS. This confirms its potential as a valuable instrument for clinicians to identify and monitor side effects in people with intellectual disabilities using psychotropic medication. We recommend the use of the Dutch version of the MEDS in both Dutch clinical practice and research for this purpose.</p>","PeriodicalId":16163,"journal":{"name":"Journal of Intellectual Disability Research","volume":" ","pages":""},"PeriodicalIF":2.1,"publicationDate":"2025-05-31","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144191898","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":"Prevalence and Incidence of Cardiovascular Disease in Adults With Intellectual Disabilities: A Systematic Review.","authors":"Marleen J de Leeuw, Thessa I M Hilgenkamp, Dederieke A M Maes-Festen, Patrick J E Bindels, Roy G Elbers, Alyt Oppewal","doi":"10.1111/jir.13254","DOIUrl":"https://doi.org/10.1111/jir.13254","url":null,"abstract":"<p><strong>Background: </strong>Given the high risk of cardiovascular diseases (CVD) in adults with intellectual disabilities (ID), there is a strong need for accurate understanding on CVD prevalence and incidence in this population. This information is important to ensure optimal care and resource allocation. However, systematic reviews on this topic are limited. Therefore, this systematic review aimed to provide a comprehensive synthesis of studies on the prevalence and incidence of CVD in adults with ID, including subgroup data.</p><p><strong>Method: </strong>We performed a systematic search in Embase, Medline ALL, Web of Science, Cochrane Central, PsycINFO and Google Scholar up to 21 January 2025, including peer-reviewed articles on CVD prevalence or incidence in adults with ID. Article screening and data extraction were independently performed by two researchers. Data were synthesised by CVD diagnosis. When available, data were reported separately for different subgroups. The methodological quality was assessed by two independent researchers. This review followed the PRISMA guidelines.</p><p><strong>Results: </strong>In 55 articles, prevalence and incidence rates were identified for coronary artery disease (prev 0%-12.9%; inc 2.0-2.8 per 1000py), myocardial infarction (prev 0%-7.9%; inc 0.3-2.8 per 1000py), heart failure (prev 0.8%-18.6%; inc 12.5 per 1000py), cerebrovascular disease (prev 0.7%-15.0%; inc 2.55 per 1000py), stroke (prev 1.3%-17.2%; inc 2.7-3.2 per 1000py), peripheral arterial disease (prev 0.4%-20.7%; inc 1.1 per 1000py), venous thrombosis (prev 0.6%-12.4%; inc 0.8-4.1 per 1000py) and atrial fibrillation (prev 0.8%-6.3%). Subgroup data have been reported based on age, sex, level of ID, aetiology of ID, living circumstances, CVD risk factors, data collection methods and source populations. Overall, higher prevalence and incidence rates were reported in older people and in studies that used physical measurements for diagnosis.</p><p><strong>Conclusions: </strong>Due to variability in methodological quality, clinical characteristics and high statistical heterogeneity, drawing conclusions about CVD prevalence and incidence in adults with ID is challenging. Therefore, the subgroup data presented in this review are valuable for identifying rates within specific subgroups. Longitudinal studies along with research employing valid and reliable data collection methods (preferably objective measurements) aligned with studies in the general population, clear reporting of individual CVD diagnoses and subgroup analyses will offer valuable additional insights in future research.</p>","PeriodicalId":16163,"journal":{"name":"Journal of Intellectual Disability Research","volume":" ","pages":""},"PeriodicalIF":2.1,"publicationDate":"2025-05-28","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144158199","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":"Fracture Characteristics Among Adults With Intellectual Disabilities and Autism Spectrum Disorders to Inform Fracture Prevention Strategies: A Descriptive Study.","authors":"Sahil A Jha, Shannen M Bolde, Edward A Hurvitz, Michelle S Caird, Daniel G Whitney","doi":"10.1111/jir.13255","DOIUrl":"https://doi.org/10.1111/jir.13255","url":null,"abstract":"<p><strong>Background: </strong>Adults with intellectual disabilities (ID) and autism spectrum disorders (ASD) have a higher risk of fracture; yet little is known about key fracture characteristics that may inform fracture prevention efforts. The objective was to describe the reported activities that lead to a fracture event, the energy of fractures (e.g., low-energy such as fragility fractures and high-energy such as fractures from a motor vehicle accident) and the location of fractures for adults with ID and ASD.</p><p><strong>Methods: </strong>In this retrospective cohort study from the United States, medical records from a single clinical site were abstracted to gather information on fracture characteristics (i.e., fracture location, energy of fracture and activities that lead to the fracture event) from adults ≥ 18 years old with intellectual disabilities (ID) and/or autism spectrum disorders (ASD) that sustained ≥ 1 fracture between 1 November 2012 and 2 November 2021. The fracture characteristics were described for the entire cohort and by the following subgroups: ID only, ASD only and ID + ASD.</p><p><strong>Results: </strong>Of the 126 adults with ID and/or ASD, there were a total of 147 fractures for analysis: 84.9% had one fracture, 13.5% had two fractures and 1.6% had three fractures. For the entire cohort, 32.0% were defined as high-energy fractures, but this varied by subgroup: 24.1% for ID only (n = 69 participants, n = 87 fractures), 50.0% for ASD only (n = 35 participants, n = 36 fractures) and 33.3% for ID + ASD (n = 22 participants, n = 24 fractures). The remaining fractures were defined as low-energy or unknown energy. The most common activities that lead to a fracture event were broadly categorised as 'low-impact falls, unwitnessed falls, transfers' for ID only (47.1%), ASD only (27.8%) and ID + ASD (41.7%). The most common skeletal region of fractures occurred in the lower extremities for ID only (42.5%) and ID + ASD (50.0%) and in the upper extremities for ASD only (33.3%).</p><p><strong>Conclusions: </strong>Despite the age being 18 years and older (i.e., not exclusively elderly), most fractures were considered to be low-energy and occurred in the extremities, but this varied by subgroup. This study identified the activities that led to a fracture event, which may inform fracture prevention efforts such as adjunct therapies.</p>","PeriodicalId":16163,"journal":{"name":"Journal of Intellectual Disability Research","volume":" ","pages":""},"PeriodicalIF":2.1,"publicationDate":"2025-05-21","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144119622","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":"Accelerometer Output and Oxygen Uptake in Adults With and Without Down Syndrome: METs vs. Percent VO_2Reserve.","authors":"S Agiovlasitis, B K Ballenger, P Choi, M Haider, Q Du, R W Motl","doi":"10.1111/jir.13253","DOIUrl":"https://doi.org/10.1111/jir.13253","url":null,"abstract":"<p><strong>Background: </strong>The estimation of physical activity (PA) and sedentary behaviour with accelerometers is typically based on the relationship between accelerometer output and metabolic equivalents (METs)-an index of PA intensity. But for adults with Down syndrome (DS), PA intensity may be better reflected in the percent oxygen uptake reserve (%VO_2Reserve), as it accounts for their lower aerobic fitness. This study examined if the relationship between accelerometer output and METs or %VO_2Reserve across various PAs and sedentary behaviours differs between adults with and without DS.</p><p><strong>Methods: </strong>Forty-one adults with DS (age 35 ± 9 years; 18 women) and 41 adults without DS (age 24 ± 5 years; 18 women) performed 17 tasks of varying intensity. We estimated aerobic fitness with a submaximal treadmill test. We measured oxygen uptake with portable calorimetry and expressed it as METs and %VO_2Reserve. Output from triaxial accelerometers on the nondominant hip and wrist was determined as Vector Magnitude (VM). We used multilevel modelling to evaluate the relationships of METs or %VO_2Reserve with VM, controlling for body mass index (BMI) and age.</p><p><strong>Results: </strong>For the hip accelerometer, VM and the group-by-VM interaction significantly predicted METs (p < 0.001; conditional R² = 0.82), but between-group differences were small. For the wrist accelerometer, VM and age significantly predicted METs (p < 0.035; conditional R² = 0.76). For both the hip and the wrist accelerometer, VM, the group-by-VM interaction and BMI significantly predicted %VO_2Reserve (p ≤ 0.047; conditional R² = 0.82 and 0.74, respectively). Between-group differences in the response to VM were larger for VO_2Reserve than METs models.</p><p><strong>Conclusion: </strong>The relationship between accelerometer output and %VO_2Reserve across PAs and sedentary behaviours is different between adults with and without DS. Calibrating accelerometer output against %VO_2Reserve may be preferable to METs.</p>","PeriodicalId":16163,"journal":{"name":"Journal of Intellectual Disability Research","volume":" ","pages":""},"PeriodicalIF":2.1,"publicationDate":"2025-05-20","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144110793","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":"Judging Offenders With Intellectual Disabilities: Systematic Review of Criminal Justice System Professionals' Expressed Views and Attitudes Towards Offenders With Intellectual Disabilities.","authors":"Georgia Powell, Kate Blake-Holmes, Adela Petrache, Rebecca Turrell, Peter Beazley","doi":"10.1111/jir.13252","DOIUrl":"https://doi.org/10.1111/jir.13252","url":null,"abstract":"<p><strong>Background: </strong>The diagnosis of an intellectual disability is suggested to have particularly stigmatising connotations, particularly within the criminal justice system (CJS). This paper aims to synthesise qualitative studies investigating the attitudes of CJS professionals to people with intellectual disabilities (PWID), specifically offenders with intellectual disabilities, and to appraise their methodological quality.</p><p><strong>Methods: </strong>A systematic search was conducted using PsychINFO, Web of Science, MEDLINE, EMBASE, CINAHL Complete and EThOS databases. Articles were screened for inclusion by title, abstract and full text to ensure predefined inclusion criteria were met. Individual study quality was rated using the 10-item Critical Appraisal Skills Programme (CASP) checklist, with the addition of an eleventh item to capture included studies' theoretical underpinnings and optimise the value of the quality appraisal. Thematic synthesis was then used to explore and synthesise the findings of the included studies.</p><p><strong>Results: </strong>Ten papers were included in the review, spanning 766 participants. Studies included utilised mixed methods surveys (n = 3), qualitative surveys (n = 1), semistructured interviews (n = 3), semistructured focus groups (n = 1), unstructured interviews (n = 1) and secondary analysis of previously collected research data (n = 1). Methodological quality was broadly of a high standard; however, all included papers failed to reflect on the relationship between the researchers and participants. Five themes were identified: conflating diagnoses, perceptions of PWID as offenders, procedural issues affecting PWID, development and maintenance of perceptions, and impact of training.</p><p><strong>Conclusions: </strong>This review highlights pervasive negative perceptions of offenders with intellectual disabilities within CJS staff groups. Clinician- and system-level factors are considered in the development and maintenance of such attitudes and suggestions made for improving CJS staff perceptions and knowledge of offenders with intellectual disabilities.</p>","PeriodicalId":16163,"journal":{"name":"Journal of Intellectual Disability Research","volume":" ","pages":""},"PeriodicalIF":2.1,"publicationDate":"2025-05-19","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144101951","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":"Characterisation of Challenging Behaviours and Associated Genetic and Neurological Features in Cardiofaciocutaneous Syndrome.","authors":"Dante J Rogers, Rebekah L Hudock, Adele F Dimian, Josue Collazo-Lopez, Ryan Shanley, Elizabeth I Pierpont","doi":"10.1111/jir.13247","DOIUrl":"https://doi.org/10.1111/jir.13247","url":null,"abstract":"<p><strong>Background: </strong>Challenging behaviours such as self-injury and aggression are prevalent among individuals with intellectual disability (ID), significantly impacting quality of life. Cardiofaciocutaneous syndrome (CFCS), a rare multisystem genetic disorder caused by variants in the BRAF, MAP2K1, MAP2K2, or KRAS genes, commonly presents with ID and other neurobehavioural features. To inform effective clinical management, we aimed to characterise and quantify challenging and repetitive behaviours in CFCS, identify functions that may maintain the behaviours, and examine associations with genotype and neurological comorbidities.</p><p><strong>Methods: </strong>In this cross-sectional cohort study, caregivers of 61 individuals with CFCS (mean age = 14.2 years; 61% female) completed an electronic survey to capture information regarding demographics, adaptive skills, and neurological history. Genotype was determined from molecular genetic testing results. The frequency, severity, topography, and function of challenging behaviours were assessed with behaviour questionnaires validated for children and adults with developmental disabilities. We evaluated trends using descriptive analyses and examined mean differences across age, genotype, and neurological variables.</p><p><strong>Results: </strong>The cohort consisted primarily of individuals with BRAF variants (62%), followed by MAP2K1 (28%) and MAP2K2 (10%) variants. Prevalence of challenging behaviour was high (77%), and self-injurious and aggressive behaviours were most frequent and severe among adolescents with CFCS relative to younger children or adults. Escape (seeking to avoid an unwanted situation or task) was the most endorsed behavioural function to maintain self-injurious and aggressive/destructive behaviours. BRAF gene variants were associated with the most frequent and variable challenging behaviours, followed by MAP2K1, and then MAP2K2. Challenging and repetitive behaviours were most prevalent among individuals with moderate adaptive functioning, clinically significant sleep disturbance, higher levels of pain interference, and more substantial sensory modulation differences. Individuals with epilepsy also exhibited more frequent repetitive and self-injurious behaviours.</p><p><strong>Conclusion: </strong>Caregivers reported a high prevalence of challenging behaviours among individuals with CFCS, especially in late childhood and adolescence. Therapeutic approaches to address challenging behaviours are needed to optimally support individuals with CFCS and their caregivers.</p>","PeriodicalId":16163,"journal":{"name":"Journal of Intellectual Disability Research","volume":" ","pages":""},"PeriodicalIF":2.1,"publicationDate":"2025-05-19","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144093895","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}