Ensuring that the outcome domains proposed for use in burns research are relevant to adult burn patients:a systematic review of qualitative research evidence

Background There have been several attempts to define core outcome domains for use in research focused on adult burns. Some have been based in expert opinion, whilst others have used primary qualitative research to understand patients’ perspectives on outcomes. To date there has not been a systematic review of qualitative research in burns to identify a comprehensive list of patient-centred outcome domains. We therefore conducted a systematic review of qualitative research studies in adult burns. Methods We searched multiple databases for English-language, peer-reviewed, qualitative research papers. We used search strategies devised using the SPIDER tool for qualitative synthesis. Our review utilized an iterative three-step approach: (1) outcome-focused coding; (2) development of descriptive accounts of outcome-relevant issues; and (3) revisiting studies and the broader theoretical literature in order to frame the review findings. Results Forty-one articles were included. We categorized papers according to their primary focus. The category with the most papers was adaptation to life following burn injury (n = 13). We defined 19 outcome domains across the 41 articles: (1) sense of self; (2) emotional and psychological morbidity; (3) sensory; (4) scarring and scar characteristics; (5) impact on relationships; (6) mobility and range of joint motion; (7) work; (8) activities of daily living and self-care; (9) treatment burden; (10) engagement in activities; (11) wound healing and infection; (12) other physical manifestations; (13) financial impact; (14) impact on spouses and family members; (15) analgesia and side effects; (16) cognitive skills; (17) length of hospital stay; (18) access to healthcare; and (19) speech and communication. We suggest that sense of self is a core concern for patients that, to date, has not been clearly conceptualized in the burns outcome domain literature. Conclusions This outcome domain framework identifies domains that are not covered in previous attempts to outline core outcome domains for adult burn research. It does so with reference to existing theoretical perspectives from the sociology and psychology of medicine. We propose that this framework can be used as a basis to ensure that outcome assessment is patient-centred. Sense of self requires further consideration as a core outcome domain

Efficacy of a self-help parenting intervention for parents of children with attention deficit hyperactivity disorder in adjunct to usual treatment—Small-scale randomized controlled trial

Background: Multimodal intervention incorporating psychosocial intervention and medication is recommended for school-aged children with attention deficit hyperactivity disorder (ADHD). This randomized controlled trial (RCT) investigates the adjunctive benefit of the self-help version of the New Forest Parenting Programme (NFPP-SH) when offered in addition to treatment as usual (TAU) compared with TAU alone. Method: Fifty-two children, receiving medication for ADHD as part of their usual care, were randomized to receive NFPP-SH + TAU or TAU alone. Results: When used in adjunct to TAU, NFPP-SH may have beneficial effects for parenting efficacy (F = 6.28, p = 0.02), child social performance in school and negative comments made by parents during a recorded speech sample. However, the self-help intervention did not have any additional effect on child behaviour. Conclusions: This study provides further support for self-help interventions as potentially low-intensity and cost-effective alternatives to therapist-led parenting interventions. The findings require replication in larger samples before any firm conclusions about adjunctive efficacy of NFPP-SH can be drawn but underline the potential for self-help within routine treatment (ClinicalTrials.gov Identifier: NCT02174952)

Anxiety and intellectual functioning in autistic children:A systematic review and meta-analysis

Autistic children experience higher levels of anxiety than their peers. Making appropriate diagnoses of anxiety disorders and providing effective treatment for these children is particularly difficult. Inconsistent evidence suggests that levels of anxiety in autistic children are related to intellectual functioning. We provide the first meta-analysis of this evidence. A systematic search identified 49 papers for review. These papers included measures of anxiety and intelligence quotient in 18,430 autistic children. Studies employing correlations showed evidence of a significant relationship between intelligence quotient and anxiety in autistic children: children with higher intelligence quotient scored higher on measures of anxiety. Studies directly comparing groups of autistic children with and without intellectual disability also supported this conclusion. Most studies employing other designs also supported this finding. Employing a quality assessment framework identified common threats to validity. Many studies used measures of anxiety that were not validated across the samples they measure. This was most notable for those autistic children with an intellectual disability. It is vital that future research determines whether the identified relationship between intelligence quotient and anxiety reflects something important in the mechanism for anxiety in autistic children, or quantifies the lack of sensitivity of our measures of anxiety across different groups

Behaviours that Challenge in SATB2-associated Syndrome: Correlates of Self-injury, Aggression and Property Destruction

SATB2-associated syndrome (SAS) is a genetic syndrome characterised by intellectual disability, severe speech delay, and palatal and dental problems. Behaviours that challenge (BtC) are reported frequently; however, there is limited research on specific forms of BtC and the correlates of these behaviours. The current study explores correlates of well-defined BtC, self-injury, aggression, and property destruction, in SAS. Eighty-one parents/caregivers of individuals with SAS (53.1% male, Mage 10.12 years) completed questionnaire measures of health, behavioural, emotional, and autism characteristics. Individuals with SAS were grouped based on caregiver responses to the presence or absence of self-injury, aggression, and property destruction on the Challenging Behaviour Questionnaire. Rates of self-injury, aggression and property destruction were 42%, 77% and 49%, respectively. Between-group comparisons were conducted to compare characteristics between behaviour groups. Significantly differing characteristics were entered into separate hierarchical logistic regressions for each form of BtC. Behavioural comparisons indicated variation in the characteristics associated with each behaviour. All hierarchical logistic regression models were significant (p <  .001): self-injury (χ2(5) = 38.46, R2 = 0.571), aggression (χ2(4) = 25.12, R2 = 0.414), property destruction (χ2(4) = 23.70, R2 = 0.346), explaining between 34.6% and 57.1% of the variance in behaviour presence. This is the first study to identify correlates of self-injury, aggression, and property destruction in SAS. Variability in the characteristics associated with each behaviour highlights the importance of specificity when examining BtC. Understanding correlates of specific forms of BtC has important implications for informing SAS-associated pathways to behavioural outcomes and the implementation of tailored behavioural interventions

Anxiety in autistic individuals who speak few or no words:A qualitative study of parental experience and anxiety management

Anxiety is a common co-occurring condition in autism and impacts quality of life of autistic individuals and their families; autistic individuals who speak few or no words represent an under-researched group. This qualitative study aimed to understand more about parental recognition and management of anxiety in autistic individuals who speak few or no words. Semi-structured interviews were conducted with parents/carers of 17 autistic individuals (m age = 14.29) recruited from an existing participant database and social media adverts. Using thematic analysis, 15 themes were placed under three a-priori grand themes: parental recognition of anxiety; parental management of anxiety; and anxiety impact on the autistic individual and their family. Due to reduced verbal language use and overlap with other behaviours, parents described difficulties recognising anxiety in their child. However, they also described use of a number of management strategies, including some which overlap with components of evidence-based interventions for emotional and behavioural problems in autistic individuals (e.g. exposure/sensory calming). Despite this, parents reported that anxiety continues to have significant impact on quality of life. The findings of this study can help to inform the development of targeted intervention and assessment measures for anxiety in autistic individuals who speak few or no words. Lay abstract: Anxiety is a common condition in autistic individuals, including those who also have an intellectual disability. Despite this, autistic individuals who have severe to profound intellectual disability, or use few or no words, are often excluded from autism research. There are also very few assessment tools and interventions with known effectiveness for autistic individuals with intellectual disability. In this study, we aimed to learn more about parent/carers experiences of recognising and managing anxiety in autistic individuals who use few or no words. We conducted semi-structured interviews with parents and carers to address three research questions: (1) what techniques and management strategies do parents describe for anxiety-related behaviour in their child; (2) how do communication difficulties impact parental understanding and management of anxiety provoking situations and behaviours; (3) what is the impact of anxiety-related behaviours on the quality of life of autistic individuals and their families? During the interviews, parents described difficulties recognising anxiety in their child, mostly due to reduced verbal language use and anxiety behaviours overlapping with other behaviours (e.g. autism characteristics). However, parents also described use of a number of management strategies, including some which overlap with components of evidence-based interventions for emotional and behavioural problems in autistic individuals (e.g. exposure/sensory calming). Despite this, parents reported that anxiety continues to have significant impact on quality of life. We will use the findings of this study to inform future research to develop assessment tools and interventions for anxiety in autistic individuals who use few or no words

The behavioral phenotype of Rubinstein–Taybi syndrome: A scoping review of the literature

Rubinstein–Taybi syndrome (RTS) is a rare genetic syndrome associated with growth delay, phenotypic facial characteristics, microcephaly, developmental delay, broad thumbs, and big toes. Most research on RTS has focused on the genotype and physical phenotype; however, several studies have described behavioral, cognitive, social, and emotional characteristics, elucidating the behavioral phenotype of RTS. The reporting of this review was informed by PRISMA guidelines. A systematic search of CINAHL, Medline, and PsychINFO was carried out in March 2021 to identify group studies describing behavioral, cognitive, emotional, psychiatric, and social characteristics in RTS. The studies were quality appraised. Characteristics reported include repetitive behavior, behaviors that challenge, intellectual disability, mental health difficulties, autism characteristics, and heightened sociability. Findings were largely consistent across studies, indicating that many characteristics are likely to form part of the behavioral phenotype of RTS. However, methodological limitations, such as a lack of appropriate comparison groups and inconsistency in measurement weaken these conclusions. There is a need for multi-disciplinary studies, combining genetic and psychological measurement expertise within single research studies. Recommendations are made for future research studies in RTS

Utilising Interview Methodology to Inform the Development of New Clinical Assessment Tools for Anxiety in Autistic Individuals Who Speak Few or no Words

Autistic individuals with intellectual disability who speak few or no words are at high risk of anxiety but are underrepresented in research. This study aimed to describe the presentation of anxiety in this population and discuss implications for the development of assessments. Interviews were conducted with 21 parents/carers of autistic individuals and nine clinicians. Data were analysed using content analysis and interpretative phenomenological analysis. Anxiety behaviours described by parents/carers included increased vocalisation, avoidance and behaviours that challenge. Changes to routine were highlighted as triggering anxiety. Clinicians discussed the importance of identifying an individual’s baseline of behaviour, knowing an individual well and ruling out other forms of distress. This study raises considerations for early identification of anxiety and for subsequent support

A novel group parenting intervention to reduce emotional and behavioural difficulties in young autistic children:protocol for the Autism Spectrum Treatment and Resilience pilot randomised controlled trial

INTRODUCTION: The majority of young autistic children display impairing emotional and behavioural difficulties that contribute to family stress. There is some evidence that behavioural parenting interventions are effective for reducing behavioural difficulties in autistic children, with less evidence assessing change in emotional difficulties. Previous trials have tended to use unblinded parent-report measures as primary outcomes and many do not employ an active control, limiting the conclusions that can be drawn. METHODS AND ANALYSIS: The Autism Spectrum Treatment and Resilience study is a pilot randomised controlled trial (RCT) testing the specific effect of a 12-week group parenting intervention (Predictive Parenting) on primary and secondary outcomes, in comparison to an attention control condition consisting of psychoeducation parent groups. Following a feasibility study to test research procedures and the interventions, the pilot RCT participants include 60 parents of autistic children aged 4-8 years who are randomised to Predictive Parenting versus the attention control. Measures are administered at baseline and post intervention to assess group differences in child and parent outcomes, costs and service use and adverse events. The primary outcome is an objective measure of child behaviours that challenge during interactions with their parent and a researcher. The trial aims to provide data on recruitment, retention, completion of measures and acceptability of the intervention and research protocol, in addition to providing a preliminary indication of potential efficacy and establishing an effect size that could be used to power a larger-scale efficacy trial. We will also provide preliminary estimates of the cost-effectiveness of the interventions. ETHICS AND DISSEMINATION: Ethical approval was granted from NHS Camden and Kings Cross Research Ethics Committee (ref: 16/LO/1769) along with NHS R&D approval from South London and Maudsley, Guy's and St Thomas', and Croydon Health Services NHS Trusts. The findings will be disseminated through publication in peer-reviewed journals and presentations at conferences. TRIAL REGISTRATION NUMBER: ISRCTN91411078

Development of the Observation Schedule for Children with Autism-Anxiety, Behaviour and Parenting (OSCA-ABP): A New Measure of Child and Parenting Behavior for Use with Young Autistic Children.

Co-occurring emotional and behavioral problems (EBPs) frequently exist in young autistic children. There is evidence based on parental report that parenting interventions reduce child EBPs. More objective measures of child EBPs should supplement parent reported outcomes in trials. We describe the development of a new measure of child and parenting behavior, the Observation Schedule for Children with Autism-Anxiety, Behaviour and Parenting (OSCA-ABP). Participants were 83 parents/carers and their 4-8-year-old autistic children. The measure demonstrated good variance and potential sensitivity to change. Child and parenting behavior were reliably coded among verbal and minimally verbal children. Associations between reports from other informants and observed behavior showed the measure had sufficient convergent validity. The measure has promise to contribute to research and clinical practice in autism mental health beyond objective measurement in trials

Prevalence of anxiety symptomatology and diagnosis in syndromic intellectual disability: a systematic review and meta-analysis

Individuals with syndromic intellectual disability are at increased risk of experiencing anxiety. Comparing prevalence estimates of anxiety will allow the identification of at-risk groups and inform causal pathways of anxiety. No known study has explored estimates of anxiety symptomatology and diagnosis, including specific anxiety profiles, across groups whilst accounting for methodological quality of studies. This systematic review and meta-analysis aimed to fill this gap. Prior to review completion, methodology and analysis plans were registered and documented in a protocol (CRD42019123561). Data from 83 papers, involving a pooled sample of 13,708 across eight syndromes were synthesised using a random effects model. Anxiety prevalence ranged from 9% (95% CI: 4-14) in Down syndrome to 73% in Rett syndrome (95% CI: 70-77). Anxiety prevalence across syndromic intellectual disability was higher than for intellectual disability of mixed aetiology and general population estimates. Substantial variability between syndromes identified groups at higher risk than others. The identification of high-risk groups is crucial for early intervention, allowing us to refine models of risk and identify divergent profiles