Narrative language abilities in adults with Down syndrome: A remote online elicitation study using the Multilingual Assessment Instrument for Narratives (MAIN)

Introduction: This research represents, to the best of our knowledge, the first attempt at assessing narrative retell remotely in people with Down syndrome and will provide valuable information on the validity and feasibility of remote online assessment with this population. Most research on language abilities in Down syndrome has focused on children and adolescents, making adults an understudied population. The present research seeks to establish a baseline of functioning for narrative language abilities in adults with Down syndrome, as part of a larger research aiming to investigate possible changes associated with aging and the emergence of Alzheimer's disease in this population. Methods: We recruited 13 adolescents and young adults with Down syndrome aged 15–33 years (mean age: 21), matched to a control group of younger typically developing children aged 4–10 years (mean age: 6) on verbal Mental Age (MA). Participants completed a picture-based story retell activity from the Multilingual Assessment Instrument for Narratives (MAIN) and a series of standardized background measures of language and cognitive ability. Results: Our analyses focused on macrostructural indices of narrative performance, narrative length and lexical diversity. Results revealed that our participants with Down syndrome were outperformed by verbal MA-matched controls on measures of story structure and story comprehension, as well as lexical diversity. No difference was found on total number of words, indicating the groups produced comparable amounts of speech despite differences in story grammar and lexis. Discussion: We interpret the results in light of previous research on macrostructural narrative performance in adults and younger adolescents with Down syndrome. Recruitment and data collection outcomes are discussed in terms of successful strategies and possible improvements. We conclude that remote online assessment of people with Down syndrome is feasible, although considerations should be made with regards to facilitating enrolment, and task engagement. Our participants demonstrated ability to engage with the experimenters over video chat and were able to complete the activities proposed mostly independently, with minimal involvement required from caregivers. Recommendations for future remote online studies involving children and people with intellectual disabilities are discussed

The effectiveness of parent-Child observation in parent-Mediated programmes for children with developmental disabilities and externalizing disorders: A systematic review and meta-analysis

This systematic review evaluated the treatment effects of communication-focused parent-mediated interventions (CF-PMT), a form of intervention that involves therapists observing parent-child interactions and giving feedback to parents on how they can practice positive parenting strategies to prevent or reduce externalizing behaviours in children with developmental disabilities. A literature search was conducted on three electronic databases. To be included in the review, studies had to: evaluate CF-PMT where therapists give feedback after observing parent-child interactions; examine changes in externalizing behaviours amongst children with any forms of developmental disability; and adopt a randomised controlled trial study design. Fifteen studies met eligible criteria for the literature review, of those, 13 studies had available data on changes in the primary (child externalizing behaviours) and secondary outcomes (parental stress, child linguistic abilities and child social responsiveness). We found significant treatment effects for CF-PMT in reducing child externalizing behaviours (d = -.60) but not for any of the secondary outcomes. A sensitivity analysis showed a small but significant treatment effect for parental stress (d = -.18). Considerable bias was observed due to the lack of available information reported by studies on aspects measured by the Mixed Methods Appraisal Tool. Overall, we found evidence to support the benefits of complex interventions which incorporate direct parent-child observations and feedback to improve behavioural outcomes amongst children with developmental disabilities

Pharmacological interventions for challenging behaviour in children with intellectual disabilities: a systematic review and meta-analysis

Background Psychotropic medications are frequently used to treat challenging behaviour in children with intellectual disabilities, despite a lack of evidence for their efficacy. This systematic review and meta-analysis aimed to determine the safety and efficacy of pharmacological interventions for challenging behaviour among children with intellectual disabilities. Methods Electronic databases were searched and supplemented with a hand search of reference lists and trial registries. Randomised controlled trials of pharmacological interventions for challenging behaviour among children with intellectual disabilities were included. Data were analysed using meta-analysis or described narratively if meta-analysis was not possible. For quality assessment, the Cochrane Risk of Bias tool and the Grading of Recommendations Assessment, Development and Evaluation (GRADE) approach were used. Results Fourteen studies including 912 participants met inclusion criteria. Antipsychotic medication reduced challenging behaviour among children with intellectual disabilities in the short-term (SMD = −1.09, p < 0.001 for risperidone; SMD = −0.64, p <0.001 for aripiprazole). However, there were significant side-effects including elevated prolactin levels (SMD = 3.22, p < 0.001) and weight gain (SMD = 0.82, p < 0.001). Evidence was inconclusive regarding the effectiveness of anticonvulsants and antioxidants for reducing challenging behaviour. The quality of all evidence was low and there were no long term follow up studies. Conclusions Antipsychotic medications appear to be effective for reducing challenging behaviour in the short-term among children with intellectual disabilities, but they carry a risk of significant side effects. Findings from this review must be interpreted with caution as studies were typically of low quality and most outcomes were based on a small number of studies. Further long-term, high-quality research is needed to determine the effectiveness and safety of psychotropic medication for reducing challenging behaviour

An exploration of lifestyle/obesity programmes for adults with intellectual disabilities through a realist lens: Impact of a ‘context, mechanism and outcome’ evaluation

Background Obesity is higher in people with intellectual disabilities. Aims There are two aims of this explorative paper. Firstly, using a realist lens, to go beyond ‘what works’ and examine the ‘context, mechanisms and outcomes’ (CMO) of lifestyle/obesity programmes for this population. Second, using a logic model framework to inform how these programmes could be implemented within practice. Method We explored six‐review papers and the individual lifestyle/obesity programmes that these papers reviewed using the CMO framework. Results There were few theoretically underpinned, multi‐component programmes that were effective in the short to long‐term and many failed to explore the ‘context and mechanisms’. We developed a logic model and engaged in two co‐production workshops to refine this model. Discussion Using a realist approach, programmes need to be underpinned by both individual and systems change theories, be multi‐component, have a closer understanding of the interplay of the ‘context and mechanisms’, and co‐designed using a logic model framework

The Association between Physical Environment and Externalising Problems in Typically Developing and Neurodiverse Children and Young People: A Narrative Review

The physical environment is of critical importance to child development. Understanding how exposure to physical environmental domains such as greenspace, urbanicity, air pollution or noise affects aggressive behaviours in typical and neurodiverse children is of particular importance given the significant long-term impact of those problems. In this narrative review, we investigated the evidence for domains of the physical environment that may ameliorate or contribute to the display of aggressive behaviours. We have considered a broad range of study designs that include typically developing and neurodiverse children and young people aged 0-18 years. We used the GRADE system to appraise the evidence. Searches were performed in eight databases in July 2020 and updated in June 2022. Additional articles were further identified by hand-searching reference lists of included papers. The protocol for the review was preregistered with PROSPERO. Results: We retrieved 7174 studies of which 67 are included in this review. The studies reported on green space, environmental noise and music, air pollution, meteorological effects, spatial density, urban or rural setting, and interior home elements (e.g., damp/sensory aspects/colour). They all used well validated parent and child reported measures of aggressive behaviour. Most of the studies were rated as having low or unclear risk of bias. As expected, noise, air pollution, urbanicity, spatial density, colour and humidity appeared to increase the display of aggressive behaviours. There was a dearth of studies on the role of the physical environment in neurodiverse children. The studies were heterogeneous and measured a range of aggressive behaviours from symptoms to full syndromes. Greenspace exposure was the most common domain studied but certainty of evidence for the association between environmental exposures and aggression problems in the child or young person was low across all domains. We found a large knowledge gap in the literature concerning neurodiverse children, which suggests that future studies should focus on these children, who are also more likely to experience adverse early life experiences including living in more deprived environments as well as being highly vulnerable to the onset of mental ill health. Such research should also aim to dis-aggregate the underlying aetiological mechanisms for environmental influences on aggression, the results of which may point to pathways for public health interventions and policy development to address inequities that can be relevant to ill health in neurodiverse young people

Identifying individuals with intellectual disability who access mental health support and are at high risk for adverse clinical outcomes: cohort study

BACKGROUND: People with intellectual disability often experience aggressive challenging behaviour and mental health issues. It can be difficult to identify those who are at higher risk of adverse clinical outcomes when in clinical care. AIMS: To characterise potential subgroups in adults with intellectual disability referred to mental health services in those presenting with aggressive behaviour or common mental disorders (CMDs). METHOD: There were 836 adults (≥18 years) with intellectual disability and a record of aggressive challenging behaviour, and 205 patients with intellectual disability and CMDs, who were seen in specialist mental health services over a 5-year period. Cluster analysis was used to define patient characteristics associated with clinical outcome. RESULTS: Distinct patient groups with differentiated profiles were observed in people with intellectual disability displaying aggressive challenging behaviour, and in those presenting with CMDs. Characteristics of the aggressive behaviour group who experienced adverse outcomes included being <30 years old, being male, more mentions of aggression and agitation in their clinical record, a diagnosis of pervasive developmental disorder and prescription of psychotropic medication. Characteristics of the CMD cluster that experienced adverse clinical outcomes were being older, being a White male, having a mild intellectual disability and physical health concerns. CONCLUSIONS: People with intellectual disability who experience adverse clinical outcomes can be identified with a cluster analysis approach of common features, but differ by clinical presentation. This could be used not only to stratify this clinically heterogeneous population in terms of response to interventions, but also improve precision in the development of tailored interventions

What works, how and in which contexts when supporting parents to implement intensive speech and language therapy at home for children with speech sound disorder? A protocol for a realist review

Introduction Speech and Language Therapists (SLTs) worldwide report challenges with providing recommended, evidence-based intervention intensity for children with speech sound disorder (SSD). Challenges such as service constraints and/or family contexts impact on access to optimal therapy intensity. Existing research indicates that empowering and training parents to deliver intervention at home, alongside SLT support, offers one possible solution to increasing the intensity of intervention children with SSD receive. Digital health could increase accessibility to intensive home-practise and help sustain engagement with therapy activities. Further exploration is needed around what makes parent-implemented interventions for children with speech sound disorder effective, for who, in which situations. This paper outlines the protocol for a realist review which aims to explore the active ingredients and contextual factors of effective digital parent-led interventions.Methods and analysis A realist review will explore the research question, following six stages. The scope of the review will be determined, and initial programme theories will be developed about what works in digital parent-implemented interventions for SSD, for whom, how, why, and in what circumstances. Relevant secondary data, identified through a formal search strategy, will be selected, appraised, analysed, and synthesised using realist principles to test and further refine the initial programme theories. This process will develop refined underpinning explanatory theories which capture the interaction between contexts, mechanisms, and outcomes of the intervention. An expert steering group will provide insight to inform explanatory theories, searches, and dissemination.Ethics and dissemination Ethical approval is not required for this review. The refined programme theories from the review will inform the next stages of a wider study. A subsequent realist evaluation will test and further refine theories with key stakeholders. Following this, the underpinning programme theory will be used to co-produce a digital tool, to support parents to deliver home-intervention alongside SLT support. <br/

Recording of intellectual disability in general hospitals in England 2006-2019: Cohort study using linked datasets

BACKGROUND: Accurate recognition and recording of intellectual disability in those who are admitted to general hospitals is necessary for making reasonable adjustments, ensuring equitable access, and monitoring quality of care. In this study, we determined the rate of recording of intellectual disability in those with the condition who were admitted to hospital and factors associated with the condition being unrecorded. METHODS AND FINDINGS: Retrospective cohort study using 2 linked datasets of routinely collected clinical data in England. We identified adults with diagnosed intellectual disability in a large secondary mental healthcare database and used general hospital records to investigate recording of intellectual disability when people were admitted to general hospitals between 2006 and 2019. Trends over time and factors associated with intellectual disability being unrecorded were investigated. We obtained data on 2,477 adults with intellectual disability who were admitted to a general hospital in England at least once during the study period (total number of admissions = 27,314; median number of admissions = 5). People with intellectual disability were accurately recorded as having the condition during 2.9% (95% CI 2.7% to 3.1%) of their admissions. Broadening the criteria to include a nonspecific code of learning difficulty increased recording to 27.7% (95% CI 27.2% to 28.3%) of all admissions. In analyses adjusted for age, sex, ethnicity, and socioeconomic deprivation, having a mild intellectual disability and being married were associated with increased odds of the intellectual disability being unrecorded in hospital records. We had no measure of quality of hospital care received and could not relate this to the presence or absence of a record of intellectual disability in the patient record. CONCLUSIONS: Recognition and recording of intellectual disability in adults admitted to English general hospitals needs to be improved. Staff awareness training, screening at the point of admission, and data sharing between health and social care services could improve care for people with intellectual disability