Autism Symptoms in Children and Young Adults With Fragile X Syndrome, Angelman Syndrome, Tuberous Sclerosis Complex, and Neurofibromatosis Type 1:A Cross-Syndrome Comparison

Objective: The etiology of autism spectrum disorder (ASD) remains unclear, due to genetic heterogeneity and heterogeneity in symptoms across individuals. This study compares ASD symptomatology between monogenetic syndromes with a high ASD prevalence, in order to reveal syndrome specific vulnerabilities and to clarify how genetic variations affect ASD symptom presentation. Methods: We assessed ASD symptom severity in children and young adults (aged 0-28 years) with Fragile X Syndrome (FXS, n = 60), Angelman Syndrome (AS, n = 91), Neurofibromatosis Type 1 (NF1, n = 279) and Tuberous Sclerosis Complex (TSC, n = 110), using the Autism Diagnostic Observation Schedule and Social Responsiveness Scale. Assessments were part of routine clinical care at the ENCORE expertise center in Rotterdam, the Netherlands. First, we compared the syndrome groups on the ASD classification prevalence and ASD severity scores. Then, we compared individuals in our syndrome groups with an ASD classification to a non-syndromic ASD group (nsASD, n = 335), on both ASD severity scores and ASD symptom profiles. Severity scores were compared using MANCOVAs with IQ and gender as covariates. Results: Overall, ASD severity scores were highest for the FXS group and lowest for the NF1 group. Compared to nsASD, individuals with an ASD classification in our syndrome groups showed less problems on the instruments' social domains. We found a relative strength in the AS group on the social cognition, communication and motivation domains and a relative challenge in creativity; a relative strength of the NF1 group on the restricted interests and repetitive behavior scale; and a relative challenge in the FXS and TSC groups on the restricted interests and repetitive behavior domain. Conclusion: The syndrome-specific strengths and challenges we found provide a frame of reference to evaluate an individual's symptoms relative to the larger syndromic population and to guide treatment decisions. Our findings support the need for personalized care and a dimensional, symptom-based diagnostic approach, in contrast to a dichotomous ASD diagnosis used as a prerequisite for access to healthcare services. Similarities in ASD symptom profiles between AS and FXS, and between NF1 and TSC may reflect similarities in their neurobiology. Deep phenotyping studies are required to link neurobiological markers to ASD symptomatology

Transition from Child and Adolescent to Adult Mental Health Services in Young People with Depression: On What Do Clinicians Base their Recommendation?

BACKGROUND: Clinicians in Child and Adolescent Mental Healthcare Services (CAMHS) face the challenge to determine who is at risk of persistence of depressive problems into adulthood and requires continued treatment after reaching the CAMHS upper age limit of care-provision. We assessed whether risk factors for persistence were related to CAMHS clinicians’ transition recommendations. METHODS: Within the wider MILESTONE cohort study, 203 CAMHS users were classified with unipolar depressive disorder by their clinician, and 185 reported clinical levels of depressive problems on the DSM-oriented Depressive Problems scale of the Achenbach Youth Self Report. Logistic regression models were fitted to both subsamples to assess the relationship between clinicians’ transition recommendations and risk factors for persistent depression. RESULTS: Only clinicianrated severity of psychopathology was related to a recommendation to continue treatment for those classified with unipolar depressive disorder (N = 203; OR = 1 45, 95% CI (1.03–2.03), p = 044) and for those with self-reported depressive problems on the Achenbach DSM-oriented Depressive Problems scale (N = 185; OR = 1 62, 95% CI (1.12–2.34), p = 012). CONCLUSION: Transition recommendations and need for continued treatment are based on clinical expertise, rather than self-reported problems and needs

The importance of clinicians' and parents' awareness of suicidal behaviour in adolescents reaching the upper age limit of their mental health services in Europe

Background: To study clinicians' and parents' awareness of suicidal behaviour in adolescents reaching the upper age limit of their Child and Adolescent Mental Health Service (CAMHS) and its association with mental health indicators, transition recommendations and mental health service (MHS) use. Methods: 763 CAMHS users from eight European countries were assessed using multi-informant and standardised assessment tools at baseline and nine months follow-up. Separate ANCOVA's and pairwise comparisons were conducted to assess whether clinicians' and parents' awareness of young people's suicidal behaviour were associated with mental health indicators, clinician's recommendations to continue treatment and MHS use at nine months follow-up. Results: 53.5 % of clinicians and 56.9 % of parents were unaware of young people's self-reported suicidal behaviour at baseline. Compared to those whose clinicians/parents were aware, unawareness was associated with a 72–80 % lower proportion of being recommended to continue treatment. Self-reported mental health problems at baseline were comparable for young people whose clinicians and parents were aware and unaware of suicidal behaviour. Clinicians' and parents' unawareness were not associated with MHS use at follow-up. Limitations: Aspects of suicidal behaviour, such as suicide ideation, -plans and -attempts, could not be distinguished. Few young people transitioned to Adult Mental Health Services (AMHS), therefore power to study factors associated with AMHS use was limited. Conclusion: Clinicians and parents are often unaware of suicidal behaviour, which decreases the likelihood of a recommendation to continue treatment, but does not seem to affect young people's MHS use or their mental health problems

Cognitive Behavioural Therapy for Anxiety Disorders in Young Children: A Dutch Open Trial of the Fun FRIENDS Program

Anxiety disorders in young children are highly prevalent and increase the risk of social, school, and familial problems, and also of psychiatric disorders in adolescence and adulthood. Nevertheless, effective interventions for this age group are lacking. One of the few available interventions is the Fun FRIENDS program. We examined whether young children with anxiety disorders showed less anxiety after participating in Fun FRIENDS. Twenty-eight clinically anxious children (4-8 years old) participated in the cognitive behavioural Fun FRIENDS program. The program consists of 12 weekly 1.5-hour sessions and was provided in groups of 3 to 5 children. At preintervention and direct postintervention, parents completed the Anxiety Disorders Interview Schedule for Children and Child Behavior Checklist. Clinically and statistically significant decreases were found in number of anxiety disorders, symptom interference, emotional and behavioural problems, internalising problems, and anxiety problems. The decrease in anxious/depressed problems and externalising problems was not significant. Furthermore, higher preintervention anxiety levels predicted more treatment progress, whereas sex and age did not. The Dutch version of Fun FRIENDS is promising in treating anxiety disorders in young children. Randomised controlled trials are needed to draw definite conclusions on the effectiveness of Fun FRIENDS in a clinical setting

Familial Aggregation of Cognitive Biases for Children with Anxiety Disorders

Previous studies described a relation between anxiety-related cognitive biases in normally developing children and parents. The current study examined the familial aggregation of cognitive biases in children with anxiety disorders (N = 55) and their parents, with possible moderators and mediators as mechanisms underlying this aggregation. Cognitive biases for children were measured by the dot-probe task for attention bias and by ambiguous stories for interpretation bias. Mothers’ (n = 50) and fathers’ (n = 30) lifetime mood and anxiety disorders were assessed, along with their attention bias (dot-probe task) and self-reported rearing styles. Results showed an association between maternal attention bias and interpretation bias of children (r = 0.31, p = 0.032). However, this association was neither moderated by maternal lifetime mood or anxiety disorders nor mediated by maternal rearing styles. The familial aggregation of maternal attention bias and children’s interpretation bias is presumably influenced by other factors than maternal mood or anxiety disorders or rearing styles

Cortical dysplasia and autistic trait severity in children with Tuberous Sclerosis Complex : a clinical epidemiological study

Tuberous Sclerosis Complex (TSC) is characterized by a high prevalence of autism spectrum disorders (ASD). Little is known about the relation between cortical dysplasia and ASD severity in TSC. We assessed ASD severity (using the Autism Diagnostic Observation Scale), tuber and radial migration line (RML) count and location, and cognitive functioning in 52 children with TSC and performed regression and mediation analyses. Tuber and RML count were strongly positively related to ASD severity. However, when correcting for cognitive functioning, the majority of associations became insignificant and only total tuber count remained associated to the severity of restricted/repetitive behaviors. Occipital RML count remained associated with overall ASD severity, and social communication/interaction deficit severity specifically. This study shows the important explanatory role of cognitive functioning in the association between cortical dysplasia and ASD severity, and the relevance of separately studying the two ASD subdomains

Online Attention Bias Modification in Combination with Cognitive-Behavioural Therapy for Children and Adolescents with Anxiety Disorders: A Randomised Controlled Trial

Attention Bias Modification (ABM) targets attention bias (AB) towards threat, which is common in youth with anxiety disorders. Previous clinical trials showed inconsistent results regarding the efficacy of ABM, and few studies have examined the effect of online ABM and its augmented effect with cognitive behavioural therapy (CBT). The aim of the current study was to examine the efficacy of online ABM combined with CBT for children and adolescents with anxiety disorders in a randomised, double-blind, placebo-controlled trial. Children (aged 8-16 years) completed nine online sessions of ABM (n = 28) or online sessions of the Attention Control Condition (ACC; n = 27) over a period of 3 weeks (modified dot-probe task with anxiety disorder-congruent stimuli), followed by CBT. Primary outcomes were clinician-reported anxiety disorder status. Secondary outcomes were patient-reported anxiety and depression symptoms and AB. Results showed a continuous decrease across time in primary and secondary outcomes (ps .50). Baseline AB and age did not moderate treatment effects. Online ABM combined with CBT does not show different efficacy compared with online ACC with CBT for children and adolescents with anxiety disorders