ABSTRACT
Diagnostic criteria for dyslexia describe specific reading difficulties, and single-deficit models, including the phonological deficit theory, have prevailed. Children seeking diagnosis, however, do not always show phonological deficits, and may present with strengths and challenges beyond reading. Through extensive neurological, neuropsychological, and academic evaluation, we describe four children with visuospatial, socio-emotional, and attention impairments and spared phonology, alongside long-standing reading difficulties. Diffusion tensor imaging revealed white matter alterations in inferior longitudinal, uncinate, and superior longitudinal fasciculi versus neurotypical children. Findings emphasize that difficulties may extend beyond reading in dyslexia and underscore the value of deep phenotyping in learning disabilities.
Subject(s)
Dyslexia , White Matter , Child , Humans , Diffusion Tensor Imaging , Phonetics , Dyslexia/psychology , ReadingABSTRACT
The purpose of this study was to examine the association between anxiety and overall academic performance (AP) in children with reading disorder (RD). This two-year longitudinal cohort study included 128 participants (aged 7-14 years) with RD. Anxiety symptoms were evaluated using the School Anxiety Scale - Teacher Report for the first year and the eight-item Spence Children's Anxiety Scale for the second year. AP was assessed by teacher ratings of progress in academic content areas, including reading, writing and math. Teachers completed evaluations every 3 months. We standardized all scores (to z scores). Multivariate linear regression models (adjusting for age, sex and Attention-Deficit/Hyperactivity Disorder symptoms) assessed the association cross-sectionally at each timepoint of survey completion. Repeated measures analysis using mixed models assessed the relationship longitudinally. Results from both univariate and multivariate analyses showed anxiety being significantly negatively associated with AP in children with RD, both cross-sectionally and longitudinally. Importantly, increased anxiety was significantly associated with reduced AP over time within an individual (adjusted ß = -0.22, p = .002). This lends support to screening for anxiety disorders in children with RD. Future research should examine the directionality of this relationship, potential mediators in the pathway and whether interventions to reduce anxiety increase AP.
Subject(s)
Academic Performance , Attention Deficit Disorder with Hyperactivity , Dyslexia , Anxiety/epidemiology , Anxiety Disorders/epidemiology , Attention Deficit Disorder with Hyperactivity/complications , Child , Cohort Studies , Humans , Longitudinal StudiesABSTRACT
[This corrects the article DOI: 10.1371/journal.pgen.1006425.].
ABSTRACT
Sexual dimorphism in common disease is pervasive, including a dramatic male preponderance in autism spectrum disorders (ASDs). Potential genetic explanations include a liability threshold model requiring increased polymorphism risk in females, sex-limited X-chromosome contribution, gene-environment interaction driven by differences in hormonal milieu, risk influenced by genes sex-differentially expressed in early brain development, or contribution from general mechanisms of sexual dimorphism shared with secondary sex characteristics. Utilizing a large single nucleotide polymorphism (SNP) dataset, we identify distinct sex-specific genome-wide significant loci. We investigate genetic hypotheses and find no evidence for increased genetic risk load in females, but evidence for sex heterogeneity on the X chromosome, and contribution of sex-heterogeneous SNPs for anthropometric traits to ASD risk. Thus, our results support pleiotropy between secondary sex characteristic determination and ASDs, providing a biological basis for sex differences in ASDs and implicating non brain-limited mechanisms.
Subject(s)
Autism Spectrum Disorder/genetics , Child Development Disorders, Pervasive/genetics , Chromosomes, Human, X/genetics , Autism Spectrum Disorder/pathology , Brain/growth & development , Brain/pathology , Child Development Disorders, Pervasive/pathology , Female , Gene-Environment Interaction , Genetic Predisposition to Disease , Genome-Wide Association Study , Genotype , Humans , Male , Polymorphism, Single Nucleotide/genetics , Sex CharacteristicsABSTRACT
BACKGROUND: Mutations in Ras/mitogen-activated protein kinase (Ras/MAPK) pathway genes lead to a class of disorders known as RASopathies, including neurofibromatosis type 1 (NF1), Noonan syndrome (NS), Costello syndrome (CS), and cardio-facio-cutaneous syndrome (CFC). Previous work has suggested potential genetic and phenotypic overlap between dysregulation of Ras/MAPK signalling and autism spectrum disorders (ASD). Although the literature offers conflicting evidence for association of NF1 and autism, there has been no systematic evaluation of autism traits in the RASopathies as a class to support a role for germline Ras/MAPK activation in ASDs. METHODS: We examined the association of autism traits with NF1, NS, CS and CFC, comparing affected probands with unaffected sibling controls and subjects with idiopathic ASDs using the qualitative Social Communication Questionnaire (SCQ) and the quantitative Social Responsiveness Scale (SRS). RESULTS: Each of the four major RASopathies showed evidence for increased qualitative and quantitative autism traits compared with sibling controls. Further, each RASopathy exhibited a distinct distribution of quantitative social impairment. Levels of social responsiveness show some evidence of correlation between sibling pairs, and autism-like impairment showed a male bias similar to idiopathic ASDs. CONCLUSIONS: Higher prevalence and severity of autism traits in RASopathies compared to unaffected siblings suggests that dysregulation of Ras/MAPK signalling during development may be implicated in ASD risk. Evidence for sex bias and potential sibling correlation suggests that autism traits in the RASopathies share characteristics with autism traits in the general population and clinical ASD population and can shed light on idiopathic ASDs.
Subject(s)
Autistic Disorder/genetics , Costello Syndrome/genetics , Ectodermal Dysplasia/genetics , Failure to Thrive/genetics , Heart Defects, Congenital/genetics , Noonan Syndrome/genetics , Quantitative Trait, Heritable , ras Proteins/genetics , Adolescent , Adult , Autistic Disorder/diagnosis , Child , Costello Syndrome/diagnosis , Diagnosis, Differential , Ectodermal Dysplasia/diagnosis , Facies , Failure to Thrive/diagnosis , Female , Heart Defects, Congenital/diagnosis , Humans , Male , Middle Aged , Mitogen-Activated Protein Kinases/metabolism , Mutation , Neuropsychological Tests , Noonan Syndrome/diagnosis , Patient Outcome Assessment , Phenotype , Prevalence , Sex Factors , Siblings , Signal Transduction , Surveys and Questionnaires , Young Adult , ras Proteins/metabolismABSTRACT
This study examined whether variations in brain development between kindergarten and Grade 3 predicted individual differences in reading ability at Grade 3. Structural MRI measurements indicated that increases in the volume of two left temporo-parietal white matter clusters are unique predictors of reading outcomes above and beyond family history, socioeconomic status, and cognitive and preliteracy measures at baseline. Using diffusion MRI, we identified the left arcuate fasciculus and superior corona radiata as key fibers within the two clusters. Bias-free regression analyses using regions of interest from prior literature revealed that volume changes in temporo-parietal white matter, together with preliteracy measures, predicted 56% of the variance in reading outcomes. Our findings demonstrate the important contribution of developmental differences in areas of left dorsal white matter, often implicated in phonological processing, as a sensitive early biomarker for later reading abilities, and by extension, reading difficulties.
Subject(s)
Child Development , Parietal Lobe/growth & development , Reading , Temporal Lobe/growth & development , White Matter/growth & development , Child , Child, Preschool , Female , Humans , Image Processing, Computer-Assisted , Magnetic Resonance Imaging , Male , Organ SizeABSTRACT
OBJECTIVE: Many geographic locations are without services and staff available to provide treatment for children with attention deficit hyperactivity disorder (ADHD). This is a randomized controlled trial to evaluate the effectiveness of group parent training on ADHD treatment delivered via videoconferencing. SUBJECTS AND METHODS: Twenty-two subjects were enrolled in the study, with 9 subjects in the videoconference session (treatment group) and 13 in the face-to-face session (control group). The parent child relationship questionnaire for child and adolescents (PCQ-CA), Vanderbilt assessment scales (parent and teacher versions), children global assessment scale, clinical global impression-severity score, clinical global impression-improvement score, and social skills rating system assessed the effectiveness of the treatment. A Likert scale evaluated parents' acceptance of the training modality. Our results showed that the parent training program significantly improved parents' disciplinary practices based on the PRQ-CA, parent ratings of ADHD, oppositional defiant disorder, and conduct disorder symptoms, and the children's global functioning. RESULTS: The treatment effects did not differ between the videoconference and face-to-face groups; however, the videoconference group evidenced statistically greater improvement on the hyperactive symptoms of Vanderbilt assessment scales. Our findings suggest that parent training through a videoconferencing modality may be as effective as face-to-face training and is well accepted by parents. CONCLUSIONS: Parent training via videoconferencing may be an important tool for addressing ADHD in geographic locations that do not have access to appropriate treatment providers.
Subject(s)
Attention Deficit Disorder with Hyperactivity/therapy , Education, Nonprofessional/methods , Parenting , Remote Consultation/methods , Videoconferencing , Adolescent , Child , Female , Humans , Male , Parent-Child Relations , Severity of Illness IndexABSTRACT
Autism spectrum disorder (ASD) is a neurodevelopmental disorder that affects 0.6%-1.7% of children. The etiology of autism is hypothesized to include both biological and environmental factors (Watts, 2008). In addition to the core symptoms of social-communication delay and restricted, repetitive interests, co-occurring irritability/aggression, hyperactivity, and insomnia negatively impact adaptive functioning and quality of life of patients and families. Despite years of effort, no pharmacologic agent has been found that targets the core symptoms of ASD. The only FDA-approved agents are risperidone and aripiprazole for agitation and irritability in ASD, not for core symptoms. Though they effectively reduce irritability/violence, they do so at the expense of problematic side effects: metabolic syndrome, elevated liver enzymes, and extrapyramidal side effects. Thus, it is not surprising that many families of children with ASD turn to nonallopathic treatment, including dietary interventions, vitamins, and immunomodulatory agents subsumed under complementary-integrative medicine (CIM). Per recent studies, 27% to 88% of families report using a CIM treatment. In an extensive population-based survey of CIM, families of children with more severe ASD, comorbid irritability, GI symptoms, food allergies, seizures, and higher parental education tend to use CIM at higher rates. The perceived safety of CIM treatments as "natural treatment" over allopathic medication increases parental comfort in using these agents. The most frequently used CIM treatments include multivitamins, an elimination diet, and Methyl B12 injections. Those perceived most effective are sensory integration, melatonin, and antifungals. Practitioners working with these families should improve their knowledge about CIM as parents currently perceive little interest in and poor knowledge of CIM by physicians. This article reviews the most popular complementary treatments preferred by families with children with autism. With many of them having limited or poor quality data, clinical recommendations about the efficacy and safety of each treatment are discussed using the SECS versus RUDE criteria.
Subject(s)
Autism Spectrum Disorder , Autistic Disorder , Integrative Medicine , Child , Humans , Autism Spectrum Disorder/drug therapy , Quality of Life , Risperidone/therapeutic useABSTRACT
Importance: There is an urgent unmet need for a treatment addressing the core symptoms and associated maladaptive symptoms of autism spectrum disorder (ASD), especially in preschool populations. Objectives: To evaluate whether treatment of children with ASD aged 3 to 6 years treated with high-protease pancreatic therapy produces long- and short-term improvements in autism-associated maladaptive behaviors. Design, Setting, and Participants: This cohort study at 32 sites across the US used a double-blind parallel group, delayed-start design comprising a 2-week blinded placebo run-in, and a double-blind, randomized, placebo-controlled segment (12 weeks). Children were recruited into the study in 2015, with data collection continuing until 2021. The analyses were completed from June 2021 to February 2022. Interventions: All participants were randomly assigned to receive either 900 mg high-protease pancreatic replacement therapy or placebo with food 3 times a day for 12 weeks, followed by all receiving 900 mg high-protease pancreatic replacement therapy for 24 weeks. Main Outcomes and Measures: The primary outcome was the irritability/agitation subscale of the Aberrant Behavior Checklist (ABC-I). All potential participants were screened using the Social Communication Questionnaire (SCQ) with diagnosis confirmed by the Diagnostic and Statistical Manual of Mental Disorders (Fourth Edition, Text Revision) for ASD and the Autism Diagnostic Inventory-Revised (ADI-R). Outcomes were measured at the conclusion of the 12-week double-blind segment and at the conclusion of the 24-week open-label segment (total 36 weeks). Results: A total of 190 participants (150 male [79%]), aged 3 to 6 (mean [SD] age, 4.5 [0.8]) years were randomized. Mixed model for repeated measures analysis performed on ABC-I demonstrated statistically significant differences of -2.49 (95% CI, -4.66 to -0.32; Cohen d = 0.364; P = .03) at the 12-week timepoint and -3.07 (95% CI, -5.81 to -0.33; Cohen d = 0.516; P = .03) at 36-week timepoint. No convergence was noted. Our high-protease pancreatic replacement (CM-AT) was well tolerated with no emergent safety concerns or related serious adverse events noted. Conclusions and Relevance: This cohort study of preschool children sustained cumulative reduction in the maladaptive behavior of irritability in autism. This delayed-start analysis, used to demonstrate disease and condition modification, may prove to be an important tool to evaluate treatments for ASD. Trial Registration: ClinicalTrials.gov Identifier: NCT02410902 and NCT02649959.
Subject(s)
Autism Spectrum Disorder , Peptide Hydrolases , Child, Preschool , Humans , Male , Autism Spectrum Disorder/therapy , Checklist , Cohort Studies , Peptide Hydrolases/therapeutic use , Female , ChildABSTRACT
OBJECTIVE: To investigate whether sluggish cognitive tempo (SCT) was associated with anxiety, depression, and academic performance (AP) in children with reading disorder (RD), and whether ADHD-Inattention (ADHD-IN) moderated these relationships. METHOD: Parents and teachers of children with RD (N = 147, ages 6-18) completed evaluations of SCT, ADHD, anxiety, depression, and AP, every 3 months for 18 months. Baseline and longitudinal associations between SCT and outcomes, and effect moderation of ADHD-IN, were assessed. RESULTS: Teacher-rated SCT was positively associated with teacher-rated anxiety (p < .001) and negatively associated with AP (p < .001) cross-sectionally and longitudinally, with significant effect modification by ADHD-IN for both outcomes. SCT was not associated with depression in adjusted cross-sectional and longitudinal analyses. There were no significant findings for any parent-reported measures. CONCLUSION: SCT has negative effects on anxiety and AP in children with RD among individuals with low ADHD-IN according to teacher report. Targeted treatment of SCT may provide substantial benefits.
Subject(s)
Academic Performance , Attention Deficit Disorder with Hyperactivity , Dyslexia , Adolescent , Attention Deficit Disorder with Hyperactivity/psychology , Child , Cognition , Cross-Sectional Studies , Dyslexia/psychology , Humans , Longitudinal Studies , Sluggish Cognitive TempoABSTRACT
CONTEXT: It has been suggested that a gluten-free and casein-free (GFCF) diet may alleviate the symptoms of autism spectrum disorder (ASD) and facilitate neurodevelopment of children with ASD. Studies to date have been inconclusive. OBJECTIVE: This study aimed to evaluate (through quantitative meta-analysis) the efficacy and safety of a GFCF diet for children with ASD. To our knowledge, this is the first time such an analysis has been carried out. DATA SOURCES: Eight electronic databases were searched, from the establishment of each database up to March 27, 2020: PubMed, Web of Science, Embase (Ovid), PsycINFO (Ovid), Cochrane Library, CNKI, Wanfang, and VIP databases. DATA EXTRACTION: Two authors independently performed the data extraction and risk-of-bias assessment. DATA ANALYSIS: A quantitative meta-analysis was performed with standard procedures by using Stata SE 15 software. Within the total of 8 studies, with 297 participants, 5 studies reported significant reductions in stereotypical behaviors [standard mean difference (SMD) = -0.41, 95% confidence interval (CI): -0.68 to -0.15], and 3 studies reported improvements in cognition (SMD = -0.46, 95% CI: -0.91 to -0.01) following GFCF dietary intervention . No statistically significant changes were observed in other symptomatic categories (all P > 0.05). CONCLUSION: The current meta-analysis showed that a GFCF diet can reduce stereotypical behaviors and improve the cognition of children with ASD. Though most of the included studies were single-blind, the benefits of a GFCF diet that have been indicated are promising. Additional studies on a larger scale are warranted. SYSTEMATIC REVIEW REGISTRATION: PROSPERO registration no. CRD42020177619.
Subject(s)
Autism Spectrum Disorder , Diet, Gluten-Free , Caseins , Child , Diet, Gluten-Free/methods , Humans , Single-Blind MethodABSTRACT
Prior studies have suggested that grit and resilience predict both academic and career success. However, these qualities have not been examined in children with reading disorder (RD). We therefore investigated whether grit and resilience were associated with anxiety, depression, academic performance, and quality of life (QOL) in these students. This 3-year longitudinal cohort study included 163 participants with RD from 3 schools. Evaluations were completed by parents and/or teachers every 3 months. The Grit and Resilience Scale was adapted from the 10-item Connor-Davidson Resilience Scale and the 12-item Grit Scale. Outcome measures included anxiety (School Anxiety Scale - Teacher Report and the 8-item Spence Children's Anxiety Scale), depression (Short Mood and Feelings Questionnaire), academic performance, and QOL (Pediatric QOL Inventory 4.0). Multivariate linear regression models (adjusting for age and sex) assessed the associations at baseline. Repeated measures analysis using mixed-effects models assessed the relationship longitudinally. There were statistically significant associations between grit and resilience and all outcomes at baseline and over time. After adjusting for age and sex, improved grit and resilience was associated with decreased anxiety (ß = - 0.4, p < 0.001) and improved academic performance (ß = 0.5, p < 0.001) when grit and resilience was measured by teachers, as well as decreased depression (ß = - 0.3, p < 0.001) and improved QOL (ß = 0.6, p < 0.001) when grit and resilience was measured by parents. Grit and resilience are significantly related to mental health, academic performance, and QOL in children with RD. This suggests that interventions to improve grit and resilience may lead to positive benefits.
Subject(s)
Dyslexia , Quality of Life , Child , Cohort Studies , Humans , Longitudinal Studies , ReadingABSTRACT
Parents of young children diagnosed with autism spectrum disorder (ASD) desperately search for a cure for their child's disorder. Depending on their child's severity or lack of progress, parents shift from looking for a cure to finding arrangements where their child can live a fulfilling life as an adult when they are less involved and less able to help. Sadly, they too often find that the resources they have fought for their children to receive disappear when their youngsters leave school.
Subject(s)
Autism Spectrum Disorder , Adult , Autism Spectrum Disorder/therapy , Child , Child, Preschool , Family , Humans , Parents , SchoolsABSTRACT
Rigid, repetitive behaviors (RRBs) as a part of autism spectrum disorder (ASD) are perplexing and challenging to treat effectively. First, how is it that they associate with deficits in social communication and social interaction to create a strong symptomatic association for the DSM-5 diagnosis of ASD?1 Deficits in social communication make sense in how they define a developmental disorder, but what is the mechanism by which social interaction and RRBs connect to define the classification of ASD? Perhaps it is that, rather than by neurotransmitters or linked structural brain regions, ASD is a neural systems condition mediated by abnormalities in regionally distributed cortical networks.2.
Subject(s)
Autism Spectrum Disorder , Brain , Cognition , Communication , Humans , Social BehaviorABSTRACT
Participants with autism spectrum disorder (ASD) (n = 121, mean [SD] age: 14.6 [8.0] years) and typically developing (TD) controls (n = 40, 16.4 [13.3] years) were presented with a series of videos representing biological motion on one side of a computer monitor screen and non-biological motion on the other, while their eye movements were recorded. As predicted, participants with ASD spent less overall time looking at presented stimuli than TD participants (P < 10-3) and showed less preference for biological motion (P < 10-5). Participants with ASD also had greater average latencies than TD participants of the first fixation on both biological (P < 0.01) and non-biological motion (P < 0.02). Findings suggest that individuals with ASD differ from TD individuals on multiple properties of eye movements and biological motion preference.
Subject(s)
Attention/physiology , Autism Spectrum Disorder/physiopathology , Autism Spectrum Disorder/psychology , Eye Movements , Motion Perception , Adolescent , Adult , Child , Eye-Tracking Technology , Female , Fixation, Ocular , Humans , Male , Middle Aged , Photic Stimulation , Prospective Studies , Task Performance and Analysis , Videotape Recording , Young AdultABSTRACT
Dyslexia is a neurodevelopmental disorder mainly defined by reading difficulties. During reading, individuals with dyslexia exhibit hypoactivity in left-lateralized language systems. Lower activity in one brain circuit can be accompanied by greater activity in another, and, here, we examined whether right-hemisphere-based emotional reactivity may be elevated in dyslexia. We measured emotional reactivity (i.e., facial behavior, physiological activity, and subjective experience) in 54 children ages 7-12 with (n = 32) and without (n = 22) dyslexia while they viewed emotion-inducing film clips. Participants also underwent task-free functional magnetic resonance imaging. Parents of children with dyslexia completed the Behavior Assessment System for Children, which assesses real-world behavior. During film viewing, children with dyslexia exhibited significantly greater reactivity in emotional facial behavior, skin conductance level, and respiration rate than those without dyslexia. Across the sample, greater emotional facial behavior correlated with stronger connectivity between right ventral anterior insula and right pregenual anterior cingulate cortex (pFWE<.05), key salience network hubs. In children with dyslexia, greater emotional facial behavior related to better real-world social skills and higher anxiety and depression. Our findings suggest there is heightened visceromotor emotional reactivity in dyslexia, which may lead to interpersonal strengths as well as affective vulnerabilities.
Subject(s)
Dyslexia , Brain Mapping , Child , Emotions , Humans , Language , Magnetic Resonance Imaging , ReadingABSTRACT
Prior studies have documented a lower quality of life (QOL) in children with autism spectrum disorder (ASD) compared to typically developing peers, but few studies have examined the trajectory of QOL over time in the same population. We conducted a 2-year cohort study in 29 children attending a specialized school for ASD with quarterly measures of parent-rated QOL as well as parent and teacher measures of behavior and social skills to determine the trajectory of change in QOL and predictors of change. The average change in QOL was constant (no change over time), but there was substantial variation with some students showing significant gains and others showing declines. Exploratory analyses revealed that improvements in behavior and social skills were greater (nonsignificantly) among children with improvements in QOL. Children with improved QOL were also younger and had a lower initial symptom burden. This study suggests that early intervention programs that provide social skills and behavioral management strategies may improve QOL in children with ASD. The study also highlights the need to develop and study novel, qualitative measures of QOL in this population.
Subject(s)
Autism Spectrum Disorder/physiopathology , Autism Spectrum Disorder/psychology , Autism Spectrum Disorder/rehabilitation , Education, Special , Outcome Assessment, Health Care , Quality of Life , Adolescent , Behavior Therapy , Child , Early Intervention, Educational , Female , Follow-Up Studies , Humans , Male , Schools , Social Behavior , Social SkillsABSTRACT
BACKGROUND: Diminished visual monitoring of faces and activities of others is an early feature of autism spectrum disorder (ASD). It is uncertain whether deficits in activity monitoring, identified using a homogeneous set of stimuli, persist throughout the lifespan in ASD, and thus, whether they could serve as a biological indicator ("biomarker") of ASD. We investigated differences in visual attention during activity monitoring in children and adult participants with autism compared to a control group of participants without autism. METHODS: Eye movements of participants with autism (n = 122; mean age [SD] = 14.5 [8.0] years) and typically developing (TD) controls (n = 40, age = 16.4 [13.3] years) were recorded while they viewed a series of videos depicting two female actors conversing while interacting with their hands over a shared task. Actors either continuously focused their gaze on each other's face (mutual gaze) or on the shared activity area (shared focus). Mean percentage looking time was computed for the activity area, actors' heads, and their bodies. RESULTS: Compared to TD participants, participants with ASD looked longer at the activity area (mean % looking time: 58.5% vs. 53.8%, p < 0.005) but less at the heads (15.2% vs. 23.7%, p < 0.0001). Additionally, within-group differences in looking time were observed between the mutual gaze and shared focus conditions in both participants without ASD (activity: Δ = - 6.4%, p < 0.004; heads: Δ = + 3.5%, p < 0.02) and participants with ASD (bodies: Δ = + 1.6%, p < 0.002). LIMITATIONS: The TD participants were not as well characterized as the participants with ASD. Inclusion criteria regarding the cognitive ability [intelligence quotient (IQ) > 60] limited the ability to include individuals with substantial intellectual disability. CONCLUSIONS: Differences in attention to faces could constitute a feature discriminative between individuals with and without ASD across the lifespan, whereas between-group differences in looking at activities may shift with development. These findings may have applications in the search for underlying biological indicators specific to ASD. Trial registration ClinicalTrials.gov identifier NCT02668991.
Subject(s)
Attention/physiology , Autism Spectrum Disorder/psychology , Social Behavior , Adolescent , Adult , Age Factors , Child , Female , Humans , Male , Middle Aged , Photic Stimulation , Task Performance and Analysis , Young AdultABSTRACT
OBJECTIVE: The relationship between sleep (caregiver-reported and actigraphy-measured) and other caregiver-reported behaviors in children and adults with autism spectrum disorder (ASD) was examined, including the use of machine learning to identify sleep variables important in predicting anxiety in ASD. METHODS: Caregivers of ASD (n = 144) and typically developing (TD) (n = 41) participants reported on sleep and other behaviors. ASD participants wore an actigraphy device at nighttime during an 8 or 10-week non-interventional study. Mean and variability of actigraphy measures for ASD participants in the week preceding midpoint and endpoint were calculated and compared with caregiver-reported and clinician-reported symptoms using a mixed effects model. An elastic-net model was developed to examine which sleep measures may drive prediction of anxiety. RESULTS: Prevalence of caregiver-reported sleep difficulties in ASD was approximately 70% and correlated significantly (p < 0.05) with sleep efficiency measured by actigraphy. Mean and variability of actigraphy measures like sleep efficiency and number of awakenings were related significantly (p < 0.05) to ASD symptom severity, hyperactivity and anxiety. In the elastic net model, caregiver-reported sleep, and variability of sleep efficiency and awakenings were amongst the important predictors of anxiety. CONCLUSION: Caregivers report problems with sleep in the majority of children and adults with ASD. Reported problems and actigraphy measures of sleep, particularly variability, are related to parent reported behaviors. Measuring variability in sleep may prove useful in understanding the relationship between sleep problems and behavior in individuals with ASD. These findings may have implications for both intervention and monitoring outcomes in ASD.
ABSTRACT
There is a need for measures to track symptom change in autism spectrum disorder (ASD). We conducted a validation study on a revised version of the Autism Behavior Inventory (ABI), and a short form (ABI-S). Caregivers of individuals (6-54 years) with confirmed diagnoses of ASD (N = 144) completed the ABI and other rating scales at 4 time points. Scale consistency for each domain, 3-5 day test-retest reliability, and construct validity, determined by comparison to pre-specified scales, were all good. Change in the ABI was congruent with changes in other instruments. Collectively, results suggest incipient suitability of the ABI as a measure of changes in core and associated symptoms of ASD.Trial Registration NCT02299700.