ABSTRACT
BACKGROUND: Youth with autism spectrum disorder (ASD) experience high rates (approximately 50-79%) of comorbid anxiety problems. Given the significant interference and distress that excessive anxiety can cause, evidence-based intervention is necessary in order to reduce long-term negative effects. Cognitive behavioral therapy (CBT) has demonstrated efficacy for treating anxiety disorders across the lifespan, both in individual and group formats. Recently, modified CBT programs for youth with ASD have been developed, showing positive outcomes. To date, these modified CBT programs have primarily been evaluated in controlled research settings. METHODS: The current community effectiveness study investigated the effectiveness of a modified group CBT program (Facing Your Fears) delivered in a tertiary care hospital and across six community-based agencies providing services for youth with ASD. Data were collected over six years (N = 105 youth with ASD; ages 6-15 years). RESULTS: Hospital and community samples did not differ significantly, except in terms of age (hospital M = 10.08 years; community M = 10.87 years). Results indicated significant improvements in anxiety levels from baseline to post-treatment across measures, with medium effect sizes. An attempt to uncover individual characteristics that predict response to treatment was unsuccessful. CONCLUSIONS: Overall, this study demonstrated that community implementation of a modified group CBT program for youth with ASD is feasible and effective for treating elevated anxiety.
Subject(s)
Anxiety/psychology , Anxiety/therapy , Autism Spectrum Disorder/psychology , Autism Spectrum Disorder/therapy , Cognitive Behavioral Therapy , Community Mental Health Services , Psychotherapy, Group , Adolescent , Analysis of Variance , Anxiety/diagnosis , Applied Behavior Analysis , Autism Spectrum Disorder/diagnosis , Child , Cognitive Behavioral Therapy/methods , Community Mental Health Services/methods , Female , Humans , Male , Prognosis , Psychotherapy, Group/methods , Symptom Assessment , Treatment OutcomeABSTRACT
De novo loss-of-function (LoF) variants in the KMT2A gene are associated with Wiedemann-Steiner Syndrome (WSS). Recently, de novo KMT2A variants have been identified in sequencing studies of cohorts of individuals with neurodevelopmental disorders (NDDs). However, most of these studies lack the detailed clinical information required to determine whether those individuals have isolated NDDs or WSS (i.e. syndromic NDDs). We performed thorough clinical and neurodevelopmental phenotyping on six individuals with de novo KMT2A variants. From these data, we found that all six patients met clinical criteria for WSS and we further define the neurodevelopmental phenotypes associated with KMT2A variants and WSS. In particular, we identified a subtype of Autism Spectrum Disorder (ASD) in five individuals, characterized by marked rigid, repetitive and inflexible behaviours, emotional dysregulation, externalizing behaviours, but relative social motivation. To further explore the clinical spectrum associated with KMT2A variants, we also conducted a meta-analysis of individuals with KMT2A variants reported in the published literature. We found that de novo LoF or missense variants in KMT2A were significantly more prevalent than predicted by a previously established statistical model of de novo mutation rate for KMT2A. Our genotype-phenotype findings better define the clinical spectrum associated with KMT2A variants and suggest that individuals with de novo LoF and missense variants likely have a clinically unrecognized diagnosis of WSS, rather than isolated NDD or ASD alone. This highlights the importance of a clinical genetic and neurodevelopmental assessment for individuals with such variants in KMT2A.
