Search

Quality of life is widely acknowledged as one of the most important outcomes in autism research, but few measures of this construct have been validated for use in autistic people. The goal of the current study was to examine the psychometric properties of the Patient-Reported Outcomes Measurement Information System Global–10, an established self-report measure of health-related quality of life, in a sample of 901 autistic adults (predominantly female and adult-diagnosed) recruited from the Simons Foundation Powering Autism Research for Knowledge cohort. Using an item response theory framework, we performed a comprehensive psychometric evaluation of the Patient-Reported Outcomes Measurement Information System Global–10 in this sample, examining its latent structure, differential item functioning, reliability, and construct validity. After developing an autism-specific measurement model, the Patient-Reported Outcomes Measurement Information System Global–10 demonstrated excellent psychometric properties in the current sample, including excellent model-data fit, high reliability, minimal differential item functioning across subgroups of autistic adults, and an expected pattern of correlations with external variables. Exploratory analyses indicated that lower quality of life was associated with female sex and identified as a sexual/gender minority. A free online score calculator has been created to facilitate the use and interpretation of normed Patient-Reported Outcomes Measurement Information System Global–10 general quality of life latent trait scores for clinical and research applications (available at https://asdmeasures.shinyapps.io/promis_qol ). Lay Abstract Quality of Life an outcome that both researchers and autistic advocates agree is extremely important to consider when implementing services, interventions, and supports for autistic people. However, there has been little research on the topic of how quality of life can best be measured in autistic people or whether existing quality of life questionnaires are appropriate for use in the autistic population. This study aimed to validate an established quality of life measure, the Patient-Reported Outcomes Measurement Information System Global–10, in a large sample of autistic adults recruited online. We created a new way to score the Patient-Reported Outcomes Measurement Information System Global–10 scale and generate a “General quality of life” score specific to autistic adults. This new score performed very well in this sample, showing very little measurement error and relating in expected ways to similar constructs, such as physical health and emotional distress. Exploratory analyses found that lower quality of life was associated with female sex and self-identification as a sexual or gender minority (i.e. LGBTQ + identity). These findings suggest that the new Patient-Reported Outcomes Measurement Information System Global–10 quality of life score is a reliable and valid measure of quality of life in autistic adults, although additional studies are necessary to further explore its measurement properties in other subsets of the autistic population, such as individuals with intellectual disabilities. This measure is freely available for use as an outcome in both research and clinical practice, and an online score calculator is available to support the use of this measure in real-world applications.

Autistic youth demonstrate differences in processing multisensory information, particularly in temporal processing of multisensory speech. Extensive research has identified several key brain regions for multisensory speech processing in non-autistic adults, including the superior temporal sulcus (STS) and insula, but it is unclear to what extent these regions are involved in temporal processing of multisensory speech in autistic youth. As a first step in exploring the neural substrates of multisensory temporal processing in this clinical population, we employed functional magnetic resonance imaging (fMRI) with a simultaneity-judgment audiovisual speech task. Eighteen autistic youth and a comparison group of 20 non-autistic youth matched on chronological age, biological sex, and gender participated. Results extend prior findings from studies of non-autistic adults, with non-autistic youth demonstrating responses in several similar regions as previously implicated in adult temporal processing of multisensory speech. Autistic youth demonstrated responses in fewer of the multisensory regions identified in adult studies; responses were limited to visual and motor cortices. Group responses in the middle temporal gyrus significantly interacted with age; younger autistic individuals showed reduced MTG responses whereas older individuals showed comparable MTG responses relative to non-autistic controls. Across groups, responses in the precuneus covaried with task accuracy, and anterior temporal and insula responses covaried with nonverbal IQ. These preliminary findings suggest possible differences in neural mechanisms of audiovisual processing in autistic youth while highlighting the need to consider participant characteristics in future, larger-scale studies exploring the neural basis of multisensory function in autism.

Autistic children show reduced multisensory integration of audiovisual speech stimuli in response to the McGurk illusion. Previously, it has been shown that adults can integrate sung McGurk tokens. These sung speech tokens offer more salient visual and auditory cues, in comparison to the spoken tokens, which may increase the identification and integration of visual speech cues in autistic children. Forty participants (20 autism, 20 non-autistic peers) aged 7–14 completed the study. Participants were presented with speech tokens in four modalities: auditory-only, visual-only, congruent audiovisual, and incongruent audiovisual (i.e., McGurk; auditory ‘ba’ and visual ‘ga’). Tokens were also presented in two formats: spoken and sung. Participants indicated what they perceived via a four-button response box (i.e., ‘ba’, ‘ga’, ‘da’, or ‘tha’). Accuracies and perception of the McGurk illusion were calculated for each modality and format. Analysis of visual-only identification indicated a significant main effect of format, whereby participants were more accurate in sung versus spoken trials, but no significant main effect of group or interaction effect. Analysis of the McGurk trials indicated no significant main effect of format or group and no significant interaction effect. Sung speech tokens improved identification of visual speech cues, but did not boost the integration of visual cues with heard speech across groups. Additional work is needed to determine what properties of spoken speech contributed to the observed improvement in visual accuracy and to evaluate whether more prolonged exposure to sung speech may yield effects on multisensory integration.

Background Autism spectrum disorder (ASD) affects many aspects of life, from social interactions to (multi)sensory processing. Similarly, the condition expresses at a variety of levels of description, from genetics to neural circuits and interpersonal behavior. We attempt to bridge between domains and levels of description by detailing the behavioral, electrophysiological, and putative neural network basis of peripersonal space (PPS) updating in ASD during a social context, given that the encoding of this space relies on appropriate multisensory integration, is malleable by social context, and is thought to delineate the boundary between the self and others. Methods Fifty (20 male/30 female) young adults, either diagnosed with ASD or age- and sex-matched individuals, took part in a visuotactile reaction time task indexing PPS, while high-density electroencephalography was continuously recorded. Neural network modeling was performed in silico. Results Multisensory psychophysics demonstrates that while PPS in neurotypical individuals shrinks in the presence of others—as to “give space”—this does not occur in ASD. Likewise, electroencephalography recordings suggest that multisensory integration is altered by social context in neurotypical individuals but not in individuals with ASD. Finally, a biologically plausible neural network model shows, as a proof of principle, that PPS updating may be inflexible in ASD owing to the altered excitatory/inhibitory balance that characterizes neural circuits in animal models of ASD. Conclusions Findings are conceptually in line with recent statistical inference accounts, suggesting diminished flexibility in ASD, and further these observations by suggesting within an example relevant for social cognition that such inflexibility may be due to excitatory/inhibitory imbalances.

Looking to the mouth of a talker early in life predicts expressive communication. We hypothesized that looking at a talker’s mouth may signal that infants are ready for increased supported joint engagement and that it subsequently facilitates prelinguistic vocal development and translates to broader gains in expressive communication. We tested this hypothesis in 50 infants aged 6–18 months with heightened and general population-level likelihood of autism diagnosis (Sibs-autism and Sibs-NA; respectively). We measured infants’ gaze to a speaker’s face using an eye tracking task, supported joint engagement during parent-child free play sessions, vocal complexity during a communication sample, and broader expressive communication. Looking at the mouth was indirectly associated with expressive communication via increased higher-order supported joint engagement and vocal complexity. This indirect effect did not vary according to sibling status. This study provides preliminary insights into the mechanisms by which looking at the mouth may influence expressive communication development.

Purpose: Caregivers of autistic children present with high stress levels, which have been associated with poorer child outcomes in several domains, including language development. However, prior to this study, it was unknown whether elevated caregiver stress was associated with language development in infant siblings of autistic children (Sibs-autism), who are at increased likelihood of receiving a future diagnosis of autism and/or language impairment compared to infant siblings of non-autistic children. This study explored the degree to which, as well as the mechanisms by which, caregiver stress was linked with later language outcomes of Sibs-autism and infant siblings of non-autistic children (Sibs-NA). Method: Participants were 50 infants (28 Sibs-autism; 22 Sibs-NA) aged 12–18 months at the first time point in this study (Time 1). Infants were seen again 9 months later, at 21–27 months of age (Time 2). Caregiver stress was measured via a validated self-report measure at Time 1. Caregiver language input, the putative mechanism by which caregiver stress may influence later language outcomes, was collected via two daylong recordings from digital recording (Language ENvironment Analysis) devices worn by the child at this same time point. Child language outcomes were measured via standardized and caregiver report measures at Time 2. Results: Several models testing hypothesized indirect effects of caregiver stress on later child language outcomes through caregiver language input were statistically significant. Specifically, significant indirect effects suggest that (a) caregivers with increased stress tend to speak less to their infants, and (b) this reduced language input tends to covary with reduced child language outcomes later in life for Sibs-autism and Sibs-NA. Conclusions: This study provides new insights into links between caregiver stress, caregiver language input, and language outcomes in Sibs-autism and Sibs-NA. Further work is necessary to understand how to best support caregivers and optimize the language learning environments for infants. Supplemental Material: https://doi.org/10.23641/asha.21714368

Early differences in sensory responsiveness may contribute to difficulties with communication among autistic children; however, this theory has not been longitudinally assessed in infants at increased familial versus general population-level likelihood for autism (Sibs-autism vs. Sibs-NA) using a comprehensive battery of sensory responsiveness and communication. In a sample of 40 infants (20 Sibs-autism, of whom six were later diagnosed with autism; 20 Sibs-NA), we tested (a) associations between sensory responsiveness at 12-18 months and communication 9 months later and (b) evaluated whether such associations were moderated by sibling group, autism diagnosis, or age. We found negative zero-order correlations between sensory responsiveness (i.e., caregiver reported hyperresponsiveness and hyporesponsiveness; an observational measure of hyperresponsiveness) and later communication. Additionally, caregiver reported sensory seeking was negatively associated with later expressive communication only in Sibs-NA. Limitations include our relatively small sample size of infants diagnosed with autism. Implications for future research are discussed.

Background Auditory perceptual abnormalities are common in persons on the autism spectrum. The neurophysiologic underpinnings of these differences have frequently been studied using auditory event-related potentials (ERPs) and event-related magnetic fields (ERFs). However, no study to date has quantitatively synthesized this literature to determine whether early auditory ERP/ERF latencies or amplitudes in autistic persons differ from those of typically developing control subjects. Methods We searched PubMed and ProQuest for studies comparing 1) latencies/amplitudes of P1/M50, N1b, N1c, M100, P2/M200, and/or N2 ERP/ERF components evoked by pure tones and 2) paired-click sensory gating (P1/N1b amplitude suppression) in autistic individuals and typically developing control subjects. Effects were synthesized using Bayesian 3-level meta-analysis. Results In response to pure tones, autistic individuals exhibited prolonged P1/M50 latencies (g = 0.341 [95% credible interval = 0.166, 0.546]), prolonged M100 latencies (g = 0.319 [0.093, 0.550]), reduced N1c amplitudes (g = −0.812 [−1.278, −0.187]), and reduced N2 amplitudes (g = −0.374 [−0.633, −0.179]). There were no practically significant group differences in P2/M200 latencies, N2 latencies, P1/M50 amplitudes, N1b amplitudes, M100 amplitudes, or P2/M200 amplitudes. Paired-click sensory gating was also reduced in autistic individuals (g = −0.389 [−0.619, −0.112]), although this effect was primarily driven by smaller responses to the first click stimulus. Conclusions Relative to typically developing control subjects, autistic individuals demonstrate multiple alterations in early cortical auditory processing of simple stimuli. However, most group differences were modest in size and based on small numbers of heterogeneous studies with variable quality. Future work is necessary to understand whether these neurophysiologic measures can predict clinically meaningful outcomes or serve as stratification biomarkers for the autistic population.

Purpose Hyperacusis is a complex and poorly understood auditory disorder characterized by decreased tolerance to sound at levels that would not trouble most individuals. Recently, it has been suggested that individuals who experience otalgia in response to everyday sounds (termed pain hyperacusis ) may differ clinically from those whose primary symptom is the perception of everyday sounds as excessively loud (termed loudness hyperacusis ). Despite this theoretical distinction, there have been no empirical studies directly comparing these two populations of hyperacusis patients. Method Using data from a multinational patient registry (the Coordination of Rare Diseases at Sanford Registry), we examined self-reported demographics, symptoms, comorbidity, and response to treatment in a sample of 243 adults with hyperacusis, 152 of whom were classified as having pain hyperacusis based on reported symptoms. Bayesian statistical tests were used to investigate both the presence and absence of group differences between patients with loudness and pain hyperacusis. Results Individuals with pain hyperacusis presented with a more severe clinical phenotype, reporting a higher frequency of temporary symptom exacerbations (i.e., “setbacks”), less perceived symptom improvement over time, more severe comorbid headache disorders, and reduced benefit from sound therapy. However, the two hypothesized hyperacusis subtypes exhibited more similarities than differences, with the majority of symptoms and comorbidities being equally prevalent across groups. Multiple comorbidities were commonly observed, including tinnitus, primary headache disorders, psychiatric disorders, and functional somatic syndromes. Intolerance of sensory stimuli in other modalities was also frequently reported. Conclusion Although this study provides little evidence that loudness and pain hyperacusis are pathophysiologically distinct conditions, our findings indicate that a pain-predominant phenotype may be a meaningful prognostic marker in patients with hyperacusis.

Children with autism show alterations in multisensory integration that have been theoretically and empirically linked with the core and related features of autism. It is unclear, however, to what extent multisensory integration maps onto features of autism within children with and without autism. This study, thus, evaluates relations between audiovisual integration and core and related autism features across children with and without autism. Thirty-six children reported perceptions of the McGurk illusion during a psychophysical task. Parents reported on participants' autistic features. Increased report of illusory percepts tended to covary with reduced autistic features and greater communication skill. Some relations, though, were moderated by group. This work suggests that associations between multisensory integration and higher-order skills are present, but in some instances vary according to diagnostic group.

PurposeDifferences in communication development impact long-term outcomes of children with autism. Previous research has identified factors associated with communication in children with autism, but much of the variance in communication skill remains unexplained. It has been proposed that early differences in sensory responsiveness (i.e., hyporesponsiveness, hyperresponsiveness, and sensory seeking) may produce “cascading effects” on communication. Evidence for this theory is limited, however, as relations between sensory responsiveness and communication in the earliest stages of development have not been well established. The purpose of this study was to evaluate (a) whether infants with a heightened likelihood of autism diagnosis (i.e., infants with an older sibling with autism) differ from infants at general population–level likelihood of autism (i.e., infants with an older, nonautistic sibling) on patterns of sensory responsiveness, (b) whether early sensory responsiveness is correlated with concurrent communication, and (c) whether the aforementioned between-groups differences and associations are moderated by age.MethodParticipants were 40 infants (20 infants with an older sibling with autism, 20 infants with an older, nonautistic sibling) aged 12–18 months. A series of observational and parent report measures of sensory responsiveness and communication skill were administered.ResultsGroup differences in sensory responsiveness across the 12- to 18-month period were limited (i.e., only observed for one measure of hyporesponsiveness), though selected differences in sensory responsiveness (i.e., parent-reported hyperresponsiveness and sensory seeking) emerged between groups over this developmental window. Parent-reported hyporesponsiveness was unconditionally, negatively associated with communication skills. Associations between expressive communication and (a) parent-reported sensory seeking and (b) an observational measure of hyperresponsiveness were moderated by age.ConclusionsThis study provides new insights into the nature of sensory responsiveness and theorized links with communication skill in infants at elevated and general population–level likelihood of autism diagnosis. Further work is needed to better characterize the effects of interest in a larger sample spanning a wider age range.Supplemental Materialhttps://doi.org/10.23641/asha.14515542

Background Individuals on the autism spectrum are reported to display alterations in interoception, the sense of the internal state of the body. The Interoception Sensory Questionnaire (ISQ) is a 20-item self-report measure of interoception specifically intended to measure this construct in autistic people. The psychometrics of the ISQ, however, have not previously been evaluated in a large sample of autistic individuals. Methods Using confirmatory factor analysis, we evaluated the latent structure of the ISQ in a large online sample of adults on the autism spectrum and found that the unidimensional model fit the data poorly. Using misspecification analysis to identify areas of local misfit and item response theory to investigate the appropriateness of the seven-point response scale, we removed redundant items and collapsed the response options to put forth a novel eight-item, five-response choice ISQ. Results The revised, five-response choice ISQ (ISQ-8) showed much improved fit while maintaining high internal reliability. Differential item functioning (DIF) analyses indicated that the items of the ISQ-8 were answered in comparable ways by autistic adolescents and adults and across multiple other sociodemographic groups. Limitations Our results were limited by the fact that we did not collect data for typically developing controls, preventing the analysis of DIF by diagnostic status. Additionally, while this study proposes a new 5-response scale for the ISQ-8, our data were not collected using this method; thus, the psychometric properties for the revised version of this instrument require further investigation. Conclusion The ISQ-8 shows promise as a reliable and valid measure of interoception in adolescents and adults on the autism spectrum, but additional work is needed to examine its psychometrics in this population. A free online score calculator has been created to facilitate the use of ISQ-8 latent trait scores for further studies of autistic adolescents and adults (available at https://asdmeasures.shinyapps.io/ISQ_score/).

Differences in audiovisual integration are commonly observed in autism. Temporal binding windows (TBWs) of audiovisual speech can be trained (i.e., narrowed) in non-autistic adults; this study evaluated a computer-based perceptual training in autistic youth and assessed whether treatment outcomes varied according to individual characteristics. Thirty autistic youth aged 8-21 were randomly assigned to a brief perceptual training (n = 15) or a control condition (n = 15). At post-test, the perceptual training group did not differ, on average, on TBWs for trained and untrained stimuli and perception of the McGurk illusion compared to the control group. The training benefited youth with higher language and nonverbal IQ scores; the training caused widened TBWs in youth with co-occurring cognitive and language impairments.

Interoception, the body’s perception of its own internal states, is thought to be altered in autism, though results have been inconsistent. The current study systematically reviewed and meta-analyzed the extant literature comparing interoceptive outcomes between autistic (AUT) and neurotypical (NT) individuals, determining which domains of interoception demonstrate robust between-group differences. A three-level Bayesian meta-analysis compared heartbeat counting performance, heartbeat discrimination performance, heartbeat counting confidence ratings, and self-reported interoceptive attention between AUT and NT groups (15 studies; n(AUT)=467, n(NT)=478). Autistic participants showed significantly reduced heartbeat counting performance (g=−0.333, CrI(95%) [−0.535, −0.138]) and higher confidence in their heartbeat counting abilities (g=0.430, CrI(95%) [0.123, 0.750]), but groups were equivalent on other meta-analyzed outcomes. Implications for future interoception research in autism are discussed.

Evidence-based practice (EBP) reviews abound in early childhood autism intervention research. These reviews seek to describe and evaluate the evidence supporting the use of specific educational and clinical practices, but give little attention to evaluating intervention outcomes in terms of the extent to which they reflect change that extends beyond the exact targets and contexts of intervention. We urge consideration of these outcome characteristics, which we refer to as "proximity" and "boundedness," as key criteria in evaluating and describing the scope of change effected by EBPs, and provide an overview and illustration of these concepts as they relate to early childhood autism intervention research. We hope this guidance will assist future researchers in selecting and evaluating intervention outcomes, as well as in making important summative determinations of the evidence base for this population. LAY SUMMARY: Recent reviews have come to somewhat different conclusions regarding the evidence base for interventions geared toward autistic children, perhaps because such reviews vary in the degree to which they consider the types of outcome measures used in past studies testing the effects of treatments. Here, we provide guidance regarding characteristics of outcome measures that research suggests are particularly important to consider when evaluating the extent to which an intervention constitutes "evidence-based practice."

We examined the quality of evidence supporting the effects of Naturalistic Developmental Behavioral Interventions (NBDIs) for facilitating change in young children with autism. We also investigated whether effects varied as a function of specific features of the intervention, samples, and outcomes measured. Twenty-seven studies testing the effects of NDBIs were extracted from data collected for the Autism Intervention Meta-analysis (Project AIM), a comprehensive meta-analysis of group design, nonpharmacological intervention studies for children with autism aged 0-8 years. We extracted effect sizes for 454 outcomes from these studies for use in meta-regression analyses testing associations between intervention effects and mean participant chronological age, language age, autism symptomatology, percentage of sample reported as male, cumulative intervention intensity, interventionist, outcome boundedness, outcome proximity, and risk of parent/teacher training correlated measurement error. The extant literature on NDBIs documents effects on social communication, language, play, and cognitive outcomes. However, our confidence in the positive and significant summary effects for these domains is somewhat limited by methodological concerns. Intervention effects were larger for context-bound outcomes (relative to generalized), and for proximal outcomes (relative to distal). Our results indicate that NDBIs have promise as an approach for supporting development for some, but not all of the core and related features of autism in early childhood. Confidence in summary effect estimates is limited by study quality concerns, particularly an overreliance on measures subject to high detection bias. The results of this review support the use of proximity and boundedness as indicators of the limits of intervention effects. LAY SUMMARY: Naturalistic Developmental Behavioral Interventions may increase language, social communication, play skills, and cognition in young children with autism, but these increases are largest for skills directly targeted by the intervention, and in contexts that are similar to that of the intervention. These conclusions are tempered by some concerns regarding research design across the studies that have been conducted to date. Autism Res 2021, 14: 817-834. © 2021 International Society for Autism Research and Wiley Periodicals LLC.

It has been challenging to elucidate the differences in brain structure that underlie behavioral features of autism. Prior studies have begun to identify patterns of changes in autism across multiple structural indices, including cortical thickness, local gyrification, and sulcal depth. However, common approaches to local gyrification indexing used in prior studies have been limited by low spatial resolution relative to functional brain topography. In this study, we analyze the aforementioned structural indices, utilizing a new method of local gyrification indexing that quantifies this index adaptively in relation to specific sulci/gyri, improving interpretation with respect to functional organization. Our sample included n = 115 autistic and n = 254 neurotypical participants aged 5–54, and we investigated structural patterns by group, age, and autism-related behaviors. Differing structural patterns by group emerged in many regions, with age moderating group differences particularly in frontal and limbic regions. There were also several regions, particularly in sensory areas, in which one or more of the structural indices of interest either positively or negatively covaried with autism-related behaviors. Given the advantages of this approach, future studies may benefit from its application in hypothesis-driven examinations of specific brain regions and/or longitudinal studies to assess brain development in autism.

Obtaining stable estimates of caregiver-child joint engagement states is of interest for researchers who study development and early intervention in young children with autism spectrum disorder (ASD). However, studies to date have offered little guidance on the numbers of sessions and coders necessary to obtain sufficiently stable estimates of these constructs. We used procedures derived from G theory to carry out a generalizability study, in which we partitioned error variance between two facets of our system for measuring joint engagement states: session and coder. A decision study was then conducted to determine the number of sessions and coders required to obtain g coefficients of 0.80, an a priori threshold set for acceptable stability. This process was conducted separately for 10 infant siblings of children with ASD (Sibs-ASD) and 10 infants whose older sibling did not have ASD (Sibs-TD), and for two different joint engagement states; lower- and higher-order supported joint engagement (LSJE and HSJE, respectively). Results indicated that, in the Sibs-ASD group, four sessions and one coder was required to obtain acceptably stable estimates for HSJE; only one session and one coder were required for LSJE. In the Sibs-TD group, two sessions and one coder were required for HSJE; seven sessions and two coders were required for LSJE. Implications for measurement in future research are discussed. Autism Res 2019, 12: 495-504 © 2019 International Society for Autism Research, Wiley Periodicals, Inc. LAY SUMMARY: This study offers guidance for researchers who measure joint engagement between caregivers and infants who have an older sibling with ASD, and who have older siblings who are TD.

An ever-growing literature has aimed to determine how individuals with autism spectrum disorder (ASD) differ from their typically developing (TD) peers on measures of multisensory integration (MSI) and to ascertain the degree to which differences in MSI are associated with the broad range of symptoms associated with ASD. Findings, however, have been highly variable across the studies carried out to date. The present work systematically reviews and quantitatively synthesizes the large literature on audiovisual MSI in individuals with ASD to evaluate the cumulative evidence for (a) group differences between individuals with ASD and TD peers, (b) correlations between MSI and autism symptoms in individuals with ASD and (c) study level factors that may moderate findings (i.e., explain differential effects) observed across studies. To identify eligible studies, a comprehensive search strategy was employed using the ProQuest search engine, PubMed database, forwards and backwards citation searches, direct author contact, and hand-searching of select conference proceedings. A significant between-group difference in MSI was evident in the literature, with individuals with ASD demonstrating worse audiovisual integration on average across studies compared to TD controls. This effect was moderated by mean participant age, such that between-group differences were more pronounced in younger samples. The mean correlation between MSI and autism and related symptomatology was also significant, indicating that increased audiovisual integration in individuals with ASD is associated with better language/communication abilities and/or reduced autism symptom severity in the extant literature. This effect was moderated by whether the stimuli were linguistic versus non-linguistic in nature, such that correlation magnitudes tended to be significantly greater when linguistic stimuli were utilized in the measure of MSI. Limitations and future directions for primary and meta-analytic research are discussed.

OBJECTIVE: Hyperacusis, defined as decreased tolerance to sound at levels that would not trouble most individuals, is frequently observed in individuals with autism spectrum disorder (ASD). Despite the functional impairment attributable to hyperacusis, little is known about its prevalence or natural history in the ASD population. The objective of this study was to conduct a systematic review and meta-analysis estimating the current and lifetime prevalence of hyperacusis in children, adolescents, and adults with ASD. By precisely estimating the burden of hyperacusis in the ASD population, the current study aims to enhance recognition of this particular symptom of ASD and highlight the need for additional research into the causes, prevention, and treatment of hyperacusis in persons on the spectrum. DESIGN: We searched PubMed and ProQuest to identify peer-reviewed articles published in English after January 1993. We additionally performed targeted searches of Google Scholar and the gray literature, including studies published through May 2020. Eligible studies included at least 20 individuals with diagnosed ASD of any age and reported data from which the proportion of ASD individuals with current and/or lifetime hyperacusis could be derived. In order to account for multiple prevalence estimates derived from the same samples, we utilized three-level Bayesian random-effects meta-analyses to estimate the current and lifetime prevalence of hyperacusis. Bayesian meta-regression was used to assess potential moderators of current hyperacusis prevalence. In order to reduce heterogeneity due to varying definitions of hyperacusis, we performed a sensitivity analysis on the subset of studies that ascertained hyperacusis status using the Autism Diagnostic Interview-Revised, a structured parent interview. RESULTS: A total of 7783 nonduplicate articles were screened, of which 67 were included in the review and synthesis. Hyperacusis status was ascertained in multiple ways across studies, with 60 articles employing interviews or questionnaires and 7 using behavioral observations or objective measures. The mean (range) age of samples in the included studies was 7.88 (1.00-34.89) years. The meta-analysis of interview/questionnaire measures (k((3)) = 103, n(ASD)=13093) estimated the current and lifetime prevalence of hyperacusis in ASD to be 41.42% (95% CrI [37.23, 45.84]) and 60.58% [50.37, 69.76], respectively. A sensitivity analysis restricted to prevalence estimates derived from the ADI-R (k((3)) = 25, n(ASD) = 5028) produced similar values. The estimate of current hyperacusis prevalence using objective/observational measures (k((3))= 8, n(ASD) = 488) was 27.30% [14.92, 46.31]. Heterogeneity in the full sample of interview/questionnaire measures was substantial but not significantly explained by any tested moderator. However, prevalence increased sharply with increasing age in studies using the ADI-R (BF(10) = 93.10, R(2)(Het) = 0.692). CONCLUSIONS AND RELEVANCE: In this meta-analysis, we found a high prevalence of current and lifetime hyperacusis in individuals with ASD, with a majority of individuals on the autism spectrum experiencing hyperacusis at some point in their lives. The high prevalence of hyperacusis in individuals with ASD across the lifespan highlights the need for further research on sound tolerance in this population and the development of services and/or interventions to reduce the burden of this common symptom.

© 2020 Elsevier Inc. The current study parsed out the distinct components of attention-deficit/hyperactivity disorder (ADHD) symptomatology to examine differential relations with language and social ability. Using a research domain criteria (RDoC) framework, we administered standardized tests and previously developed and validated questionnaires to assess levels of inattention and/or hyperactivity/impulsivity symptomatology, language, social responsivity and social competency in 98 young adults. Those with higher inattention and/or hyperactivity/impulsivity symptomatology had reduced language comprehension, social responsivity, and social competency. Inattention and hyperactivity/impulsivity both predicted language comprehension, but not language production. Interestingly, inattention uniquely contributed to social responsiveness and social competency, but hyperactivity/impulsivity did not. Findings suggest that inattention and/or hyperactivity/impulsivity symptoms, inattention in particular, may be especially important for social skills programs geared towards individuals with attention limitations.

Autism spectrum disorder (ASD) is a heterogenous disorder predominantly characterized by social and communicative differences, but increasingly recognized to also alter (multi)sensory function. To face the heterogeneity and ubiquity of ASD, researchers have proposed models of statistical inference operating at the level of ‘computations’. Here, we attempt to bridge both across domains – from social to sensory – and levels of description – from behavioral computations to neural ensemble activity to a biologically-plausible artificial neural network – in furthering our understanding of autism. We do so by mapping visuo-tactile peri-personal space (PPS), and examining its electroencephalography (EEG) correlates, in individuals with ASD and neurotypical individuals during both a social and non-social context given that (i) the sensory coding of PPS is well understood, (ii) this space is thought to distinguish between self and other, and (iii) PPS is known to remap during social interactions. In contrast to their neurotypical counterparts, psychophysical and EEG evidence suggested that PPS does not remap in ASD during a social context. To account for this observation, we then employed a neural network model of PPS and demonstrate that PPS remapping may be driven by changes in neural gain operating at the level of multisensory neurons. Critically, under the anomalous excitation-inhibition (E/I) regime of ASD, this gain modulation does not result in PPS resizing. Overall, our findings are in line with recent statistical inference accounts suggesting diminished flexibility in ASD, and further these accounts by demonstrating within an example relevant for social cognition that such inflexibility may be due to E/I imbalances.

Our team examined 150 reports on group-design, non-pharmacological interventions for young autistic children, to determine the prevalence of adverse event reporting. We found that only 11 studies mentioned adverse events; one indicated adverse events occurred, and an additional three indicated adverse effects occurred (i.e. adverse events that could be attributed to the intervention). We also coded reasons for participant withdrawal and found that of the 54 studies that reported reasons for withdrawal, 10 studies reported reasons that could be categorized as adverse events, 8 reported reasons that could be categorized as adverse effects, and an additional 12 studies reported reasons that were too vaguely described to determine adverse event status. We recommend that autism intervention researchers make concerted efforts to monitor, classify, and report adverse events so that practitioners, policy-makers, and families are better equipped to weigh potential benefits of interventions against potential harms. Lay abstract In this study, we looked at published research on interventions for young autistic children that did not involve administering medication. We were interested in determining how often studies reported on whether adverse events (i.e. physical or psychological distress to the participants) or adverse effects (i.e. adverse events that are thought to be caused by the intervention) had occurred. We found that of the 150 reports we examined, only 11 mentioned adverse events. One of these studies reported adverse events occurred, and three reported that adverse effects occurred. We also reviewed the studies to examine the reasons that were given to explain why any participants dropped out of the intervention (termed “withdrawal”), to determine if any of these reasons could be considered adverse events or adverse effects. Fifty-four studies described reasons for withdrawal, and 10 of these studies had reasons that could be categorized as an adverse event, 8 studies had reasons that could be categorized as an adverse effect, and an additional 12 studies had reasons that were too vaguely described to determine whether they were adverse events or not. We recommend that autism intervention researchers develop more systematic methods of looking for and reporting adverse events and effects, so that professionals and families can be better informed when choosing to enroll their autistic children in interventions.

Autism is a neurodevelopmental condition defined by differences in social communication and by the presence of restricted and repetitive patterns of behavior, interests, and activities (RRBs). Individuals with autism also commonly present with atypical patterns of sensory responsiveness (i.e., hyporesponsiveness, hyperresponsiveness, and sensory seeking), which are theorized to produce cascading effects across other domains of development. The purpose of this study was to examine differences in sensory responsiveness in children with and without autism (ages 8&ndash, 18 years), as well as relations between patterns of sensory responsiveness and core and related features of autism. Participants were 50 children with autism and 50 non-autistic peers matched on age and sex. A comprehensive clinical battery included multiple measures of sensory responsiveness, core features of autism, adaptive behavior, internalizing behaviors, cognitive ability, and language ability. Groups significantly differed on all three patterns of sensory responsiveness. Some indices of core and related autism features were robustly associated with all three patterns of sensory responsiveness (e.g., RRBs), while others were more strongly associated with discrete patterns of sensory responsiveness (i.e., internalizing problem behaviors and hyperresponsiveness, language and sensory seeking). This study extends prior work to show that differences in sensory responsiveness that are linked with core and related features of autism persist in older children and adolescents on the spectrum.

Atypical behavioral responses to environmental sounds are common in autistic children and adults, with 50-70 % of this population exhibiting decreased sound tolerance (DST) at some point in their lives. This symptom is a source of significant distress and impairment across the lifespan, contributing to anxiety, challenging behaviors, reduced community participation, and school/workplace difficulties. However, relatively little is known about its phenomenology or neurocognitive underpinnings. The present article synthesizes a large body of literature on the phenomenology and pathophysiology of DST-related conditions to generate a comprehensive theoretical account of DST in autism. Notably, we argue against conceptualizing DST as a unified construct, suggesting that it be separated into three phenomenologically distinct conditions: hyperacusis (the perception of everyday sounds as excessively loud or painful), misophonia (an acquired aversive reaction to specific sounds), and phonophobia (a specific phobia of sound), each responsible for a portion of observed DST behaviors. We further elaborate our framework by proposing preliminary neurocognitive models of hyperacusis, misophonia, and phonophobia that incorporate neurophysiologic findings from studies of autism.

The current study parsed out the distinct components of attention-deficit/hyperactivity disorder (ADHD) symptomatology to examine differential relations with language and social ability. Using a research domain criteria (RDoC) framework, we administered standardized tests and previously developed and validated questionnaires to assess levels of inattention and/or hyperactivity/impulsivity symptomatology, language, social responsivity and social competency in 98 young adults. Those with higher inattention and/or hyperactivity/impulsivity symptomatology had reduced language comprehension, social responsivity, and social competency. Inattention and hyperactivity/impulsivity both predicted language comprehension, but not language production. Interestingly, inattention uniquely contributed to social responsiveness and social competency, but hyperactivity/impulsivity did not. Findings suggest that inattention and/or hyperactivity/impulsivity symptoms, inattention in particular, may be especially important for social skills programs geared towards individuals with attention limitations.

Purpose This study synthesized effects of interventions on language outcomes of young children (ages 0–8 years) with autism and evaluated the extent to which summary effects varied by intervention, participant, and outcome characteristics. Method A subset of effect sizes gathered for a larger meta-analysis (the Autism Intervention Meta-analysis or Project AIM) examining the effects of interventions for young children with autism, which were specific to language outcomes, was analyzed. Robust variance estimation and metaregression were used to calculate summary and moderated effects while controlling for intercorrelation among outcomes within studies. Results A total of 221 outcomes were gathered from 60 studies. The summary effect of intervention on language outcomes was small but significant. Summary effects were larger for expressive and composite language outcomes compared to receptive language outcomes. Interventions implemented by clinicians, or by clinicians and caregivers together, had summary effects that were significantly larger than interventions implemented by caregivers alone. Participants' pretreatment language age equivalent scores positively and significantly moderated intervention effects, such that effects were significantly larger on average when samples of children had higher pretreatment language levels. Effects were not moderated by cumulative intervention intensity, intervention type, autism symptomatology, chronological age, or the proximity or boundedness of outcomes. Study quality concerns were apparent for a majority of included outcomes. Conclusions We found evidence that intervention can facilitate improvements in language outcomes for young children with autism. Effects were largest for expressive and composite language outcomes, for children with initially higher language abilities, and for interventions implemented by clinicians or by caregivers and clinicians combined. However, quality concerns of included studies and borderline significance of some results temper our conclusions regarding intervention effectiveness and corresponding moderators.

Background The presence, types, disclosure rates, and effects of conflicts of interest (COIs) on autism early intervention research have not previously been studied. The purpose of this study was to examine these issues. Methods This study is a secondary analysis of a comprehensive meta-analysis of all group-design, nonpharmacological early intervention autism research conducted between 1970 and 2018. We coded reports for the presence/absence of COI statements, the types of COIs that were disclosed, and for 8 types of COIs, including (a) the author developed the intervention, (b) the author is affiliated with a clinical provider, (c) the author is employed by a clinical provider, (d) the author is affiliated with an institution that trains others to use the intervention, (e) the author receives payment or royalties related to the intervention, (f) the study was funded by an intervention provider, (g) the study used a commercially available measure developed by the author, and (h) proceeds of the intervention fund the author's research. Frequencies and proportions were calculated to determine prevalence of COIs and COI disclosures. Meta-analysis was used to estimate summary effects by COI type and to determine if they were larger than for reports with no coded COIs. Results Seventy percent of reports were coded for ≥ 1 COI, but only ~ 6% of reports contained COI statements fully accounting for all coded COIs. Metaregressions did not detect significant influences of any COI type on summary effects; however, point estimates for each COI type were larger than for reports with no coded COIs. Conclusions Conflicts of interest are prevalent but under-reported in autism early intervention research. Improved reporting practices are necessary for researcher transparency and would enable more robust examination of the effects of COIs on research outcomes.

Research shows that children with autism spectrum disorder (ASD) differ in their behavioral patterns of responding to sensory stimuli (i.e., sensory responsiveness) and in various other aspects of sensory functioning relative to typical peers. This study explored relations between measures of sensory responsiveness and multisensory speech perception and integration in children with and without ASD. Participants were 8-17 year old children, 18 with ASD and 18 matched typically developing controls. Participants completed a psychophysical speech perception task, and parents reported on children's sensory responsiveness. Psychophysical measures (e.g., audiovisual accuracy, temporal binding window) were associated with patterns of sensory responsiveness (e.g., hyporesponsiveness, sensory seeking). Results indicate that differences in multisensory speech perception and integration covary with atypical patterns of sensory responsiveness.

The Short Sensory Profile (SSP) is one of the most commonly used measures of sensory features in children with autism spectrum disorder (ASD), but psychometric studies in this population are limited. Using confirmatory factor analysis, we evaluated the structural validity of the SSP subscales in ASD children. Confirmatory factor models exhibited poor fit, and a follow-up exploratory factor analysis suggested a 9-factor structure that only replicated three of the seven original subscales. Secondary analyses suggest that while reliable, the SSP total score is substantially biased by individual differences on dimensions other than the general factor. Overall, our findings discourage the use of the SSP total score and most subscale scores in children with ASD. Implications for future research are discussed.

Children's vocal development occurs in the context of reciprocal exchanges with a communication partner who models "speechlike" productions. We propose a new measure of child vocal reciprocity, which we define as the degree to which an adult vocal response increases the probability of an immediately following child vocal response. Vocal reciprocity is likely to be associated with the speechlikeness of vocal communication in young children with autism spectrum disorder (ASD). Two studies were conducted to test the utility of the new measure. The first used simulated vocal samples with randomly sequenced child and adult vocalizations to test the accuracy of the proposed index of child vocal reciprocity. The second was an empirical study of 21 children with ASD who were preverbal or in the early stages of language development. Daylong vocal samples collected in the natural environment were computer analyzed to derive the proposed index of child vocal reciprocity, which was highly stable when derived from two daylong vocal samples and was associated with speechlikeness of vocal communication. This association was significant even when controlling for chance probability of child vocalizations to adult vocal responses, probability of adult vocalizations, or probability of child vocalizations. A valid measure of children's vocal reciprocity might eventually improve our ability to predict which children are on track to develop useful speech and/or are most likely to respond to language intervention. A link to a free, publicly-available software program to derive the new measure of child vocal reciprocity is provided. Autism Res 2018, 11: 903-915. © 2018 International Society for Autism Research, Wiley Periodicals, Inc. Lay summary Children and adults often engage in back-and-forth vocal exchanges. The extent to which they do so is believed to support children's early speech and language development. Two studies tested a new measure of child vocal reciprocity using computer-generated and real-life vocal samples of young children with autism collected in natural settings. The results provide initial evidence of accuracy, test-retest reliability, and validity of the new measure of child vocal reciprocity. A sound measure of children's vocal reciprocity might improve our ability to predict which children are on track to develop useful speech and/or are most likely to respond to language intervention. A free, publicly-available software program and manuals are provided.

We examined differences between children with autism spectrum disorder and typically developing children over an 8-month period in: (a) longitudinal associations between expressive and receptive vocabulary and (b) the extent to which caregiver utterances provided within an “optimal” engagement state mediated the pathway from early expressive to later receptive vocabulary. In total, 59 children (28–53 months at Time 1) comprised the autism spectrum disorder group and 46 children (8–24 months at Time 1) comprised the typically developing group. Groups were matched on initial vocabulary sizes. Results showed that the association between early expressive and later receptive vocabulary was moderated by group. A moderated mediation effect was also found, indicating linguistic input provided within an optimal engagement state only mediated associations for the autism spectrum disorder group.

Altered patterns of sensory responsiveness are a frequently reported feature of Autism Spectrum Disorder (ASD). Younger siblings of individuals with ASD are at a greatly elevated risk of a future diagnosis of ASD, but little is known about the neural basis of sensory responsiveness patterns in this population. Younger siblings (n = 20) of children diagnosed with ASD participated in resting electroencephalography (EEG) at an age of 18 months. Data on toddlers’ sensory responsiveness were obtained using the Sensory Experiences Questionnaire. Correlations were present between hyporesponsiveness and patterns of oscillatory power, functional connectivity, and signal complexity. Our findings suggest that neural signal features hold promise for facilitating early identification and targeted remediation in young children at risk for ASD.

Intentional communication has previously been identified as a value-added predictor of expressive language in preverbal preschoolers with autism spectrum disorder. In the present study, we sought to identify value-added predictors of intentional communication. Of five theoretically-motivated putative predictors of intentional communication measured early in the study (at study entry and 4 months after), three had significant zero-order correlations with later intentional communication (12 months after study entry) and were thus added to a linear model that predicted later intentional communication scores controlling for initial intentional communication scores at study entry. After controlling for initial intentional communication, early motor imitation was the only predictor that accounted for a significant amount of variance in children's later intentional communication.

In this comprehensive systematic review and meta-analysis of group design studies of nonpharmacological early interventions designed for young children with autism spectrum disorder (ASD), we report summary effects across 7 early intervention types (behavioral, developmental, naturalistic developmental behavioral intervention [NDBI], TEACCH, sensory-based, animal-assisted, and technology-based), and 15 outcome categories indexing core and related ASD symptoms. A total of 1,615 effect sizes were gathered from 130 independent participant samples. A total of 6,240 participants, who ranged in age from 0-8 years, are represented across the studies. We synthesized effects within intervention and outcome type using a robust variance estimation approach to account for the nesting of effect sizes within studies. We also tracked study quality indicators, and report an additional set of summary effect sizes that restrict included studies to those meeting prespecified quality indicators. Finally, we conducted moderator analyses to evaluate whether summary effects across intervention types were larger for proximal as compared with distal effects, and for context-bound as compared to generalized effects. We found that when study quality indicators were not taken into account, significant positive effects were found for behavioral, developmental, and NDBI intervention types. When effect size estimation was limited to studies with randomized controlled trial (RCT) designs, evidence of positive summary effects existed only for developmental and NDBI intervention types. This was also the case when outcomes measured by parent report were excluded. Finally, when effect estimation was limited to RCT designs and to outcomes for which there was no risk of detection bias, no intervention types showed significant effects on any outcome. (PsycINFO Database Record (c) 2019 APA, all rights reserved).

Purpose We examined associations between vocal communication with canonical syllables and expressive language and then examined 2 potential alternative explanations for such associations. Method Specifically, we tested whether the associations remained when excluding canonical syllables in identifiable words and controlling for the number of communication acts. Participants included 68 preverbal or low verbal children with autism spectrum disorder ( M age = 35.26 months). Results Vocal communication with canonical syllables and expressive language were concurrently and longitudinally associated with moderate to strong ( R 2 s = .13–.70) and significant ( p s < .001) effect sizes. Even when excluding spoken words from the vocal predictor and controlling for the number of communication acts, vocal communication with canonical syllables predicted expressive language. Conclusions The findings provide increased support for measuring vocal communication with canonical syllables and for examining a causal relation between vocal communication with canonical syllables and expressive language in children with ASD who are preverbal or low verbal. In future studies, it may be unnecessary to eliminate identifiable words when measuring vocal communication in this population. Following replication, vocal communication with canonical syllables may be considered when making intervention- planning decisions.

Children with autism spectrum disorder (ASD) display differences in multisensory function as quantified by several different measures. This study estimated the stability of variables derived from commonly used measures of multisensory function in school-aged children with ASD. Participants completed: a simultaneity judgment task for audiovisual speech, tasks designed to elicit the McGurk effect, listening-in-noise tasks, electroencephalographic recordings, and eye-tracking tasks. Results indicate the stability of indices derived from tasks tapping multisensory processing is variable. These findings have important implications for measurement in future research. Averaging scores across repeated observations will often be required to obtain acceptably stable estimates and, thus, to increase the likelihood of detecting effects of interest, as it relates to multisensory processing in children with ASD.