Social COmmunication Program supported by E-health (SCOPE) for infants and toddlers at elevated likelihood of autism spectrum disorder: study design of a cluster randomized controlled trial.

BACKGROUND: Although the importance of early detection and early intervention of autism spectrum disorders (ASD) is widely recognized, multiple barriers exist in accessing early intervention services. As an alternative to these barriers, the SCOPE project presents a new, easy accessible and blended intervention called BEAR (Blended E-health for children at eArly Risk). This paper describes this BEAR intervention and study design of an ongoing two arm cluster randomized controlled trial (RCT). METHODS: BEAR (Blended E-health for children at eArly Risk) is a blended e-health intervention, based on evidence-based naturalistic developmental behavioral interventions (NDBI's) and can be offered to parents and infants/toddlers at high likelihood for ASD. During the ongoing RCT, N = 88 high risk infants and toddlers will be cluster randomized over the BEAR intervention and care-as-usual (CAU) conditions. The finalized version of the intervention protocol and study design are presented in this paper. The primary outcome measure is joint engagement measured by the Joint Engagement Rating Inventory (JERI) during videotaped parent-child interaction. Secondary outcome measures include severity of ASD symptoms, global level of adaptive functioning, parental well-being, parental skills and satisfaction with healthcare. Also, costs will be estimated from society's perspective. Assessments take place at the start of the study (T1), after eight weeks (T2) and after six months (T3) and include behavioral home observations and parental questionnaires. DISCUSSION: The SCOPE project aims to contribute to improved early identification and timely start of suitable interventions for infants and toddlers at elevated likelihood for ASD. This ongoing RCT will offer insight in the feasibility, short-term and six months effects of the innovative BEAR intervention. It is estimated that inclusion for the trial (N = 88) is completed in spring 2023. TRIAL REGISTRATION: Dutch Trial Register, NTR7695. Registered at December 17th, 2018, www.trialregister.nl .

Pivotal Response Treatment with and without robot-assistance for children with autism: a randomized controlled trial.

Pivotal response treatment (PRT) is a promising intervention focused on improving social communication skills in children with autism spectrum disorder (ASD). Since robots potentially appeal to children with ASD and may contribute to their motivation for social interaction, this exploratory randomized controlled trial (RCT) was conducted comparing PRT (PRT and robot-assisted PRT) with treatment-as-usual (TAU). Seventy-three children (PRT: n = 25; PRT + robot: n = 25; TAU: n = 23) with ASD, aged 3-8 years were assessed at baseline, after 10 and 20 weeks of intervention, and at 3-month follow-up. There were no significant group differences on parent- and teacher-rated general social-communicative skills and blindly rated global functioning directly after treatment. However, at follow-up largest gains were observed in robot-assisted PRT compared to other groups. These results suggest that robot-assistance may contribute to intervention efficacy for children with ASD when using game scenarios for robot-child interaction during multiple sessions combined with motivational components of PRT. This trial is registered at https://www.trialregister.nl/trial/4487 ; NL4487/NTR4712 (2014-08-01).

Building a theoretical framework for autism spectrum disorders screening instruments in Europe.

BACKGROUND: This study addresses the need for a theoretical base to develop more effective early autism spectrum disorders (ASD) detection tools. The structure that underlies early ASD detection is explored by evaluating the opinions of experts on ASD screening tools currently used in Europe. METHOD: A process of face and content validity was performed. First, the best constructs were selected from the relevant tests: Checklist for Early Signs of Developmental Disorders (CESDD), Checklist for Autism in Toddlers (CHAT), Early Screening of Autistic Traits Questionnaire (ESAT), Modified Checklist for Autism in Toddlers (M-CHAT), Social Communication Questionnaire (SCQ) and Communication and Symbolic Behaviour Scales Developmental Profile (CSBS-DP). The diagnostic content validity model by Fehring (1986, 1994) was adapted to make the selection. Afterwards, the items, taken from these tests, were selected to fit into each construct, using the same methodology. RESULTS: Twelve of the 18 constructs were selected by the experts and 11 items were chosen from a total of 130, reduced to eight after eliminating tautologies. CONCLUSIONS: Mapping these constructs and items on to the DSM-5 diagnostic criteria for ASD indicated good face and content validity. Results of this research will contribute to efforts to improve early ASD screening instruments and identify the key behaviours that experts in ASD see as the most relevant for early detection.

Network Structure of Autism Spectrum Disorder Behaviors and Its Evolution in Preschool Children: Insights from a New Longitudinal Network Analysis Method.

Network modeling of the social, communication and restrictive/repetitive behaviors (RRBs) included in the definition of Autism Spectrum Disorder was performed. The Autism Diagnostic Interview-Revised (ADI-R) assessed behaviors in 139 pre-school cases at two cross-sections that averaged 34.8 months apart. Cross-sectional networks were based on the correlation matrix of the ADI-R behavioral items and the "bootCross" method was developed and enabled the estimation of a longitudinal network. At both stages, RRB items/nodes formed a consistent peripheral cluster, while social and communication nodes formed a core cluster that diverged with time. These differences in the nature and evolution of the RRB and socio-communicative dimensions indicate that their inter-behavior dynamics are very different. The most central behaviors across stages are proposed as prime targets for efficient therapeutic intervention.

Pivotal Response Treatment (PRT) parent group training for young children with autism spectrum disorder: a pilot study.

Pivotal Response Treatment (PRT) is a promising intervention addressing core symptoms of autism spectrum disorder (ASD), with parent involvement as key component. Parent group-delivered PRT may be an effective treatment model, but currently the evidence is limited. Also, little attention has been paid to therapeutic involvement of multiple important contexts (e.g. home, school, community) of the young child. The current study explores a 14-week protocol of PRT parent group training (PRT-PG), complemented with individual parent-child sessions and involvement of teachers and other childcare providers. Children aged 2-6 years old with ASD and their parents (n = 20) were included. Preliminary results showed a significant increase in spontaneous initiations during a semi-structured therapist-child interaction together with widespread gains in clinical global functioning. No significant improvement on parent-rated general social-communication skills was observed. These findings justify further research on parent group delivered PRT models.

Parental Experiences with Early Identification and Initial Care for their Child with Autism: Tailored Improvement Strategies.

Whereas it is well documented how parents experience the diagnostic process of their child with autism spectrum disorder (ASD), less is known about parental experiences with the course of the early identification process and first steps in receiving care for their child with ASD symptoms. This mixed-method study investigated these experiences as well as barriers and improvement strategies regarding early detection in the Netherlands. A parental survey (N = 45) showed that, on average, initial concerns started at 22 months. A focus group (N = 10) revealed multiple barriers and proposed strategies of improvement in three domains: "Knowledge and Expertise", "Attention to Parental Needs" and "System and Organization". Strategies to improve early identification will be discussed based on parental perspectives and professional perspectives.

Early detection of young children at risk of autism spectrum disorder at well-baby clinics in the Netherlands: Perspectives of preventive care physicians.

LAY ABSTRACT: To improve early detection of autism spectrum disorder in preventive care, a Dutch guideline was developed 5 years ago. The guideline provides preventive care physicians at well-baby clinics action-oriented advice and describes a step-by-step approach for children identified at an increased risk for autism spectrum disorder during general healthcare surveillance. The present qualitative study evaluated adherence to the guideline and studied barriers regarding early detection of autism spectrum disorder at well-baby clinics. Interviews were undertaken with 12 preventive care physicians (one representative per province). It was found that the vast majority of participants did not follow-up general surveillance with an autism spectrum disorder -specific screener as prescribed by the guideline. Six barriers (limited knowledge about autism spectrum disorder symptoms in infant and toddlerhood, professional attitude toward early detection, problems in discussing initial worries with parents, limited use of screening instruments, perceptions toward cultural and language differences and constraints regarding availability of healthcare services) were found. The results of this study highlight the importance of an integrative approach, raising awareness of the benefits regarding early detection of autism spectrum disorder in preventive care, the need of continuous investment in easy and accessible training and active screening, and a closer collaboration between preventive care organizations and autism spectrum disorder experts.

Advancing early detection of autism spectrum disorder by applying an integrated two-stage screening approach.

BACKGROUND: Few field trials exist on the impact of implementing guidelines for the early detection of autism spectrum disorders (ASD). The aims of the present study were to develop and evaluate a clinically relevant integrated early detection programme based on the two-stage screening approach of Filipek et al. (1999), and to expand the evidence base for this approach. METHODS: The integrated early detection programme encompassed: 1) training relevant professionals to recognise early signs of autism and to use the Early Screening of Autistic Traits Questionnaire (ESAT; Dietz, Swinkels et al., 2006; Swinkels, van Daalen, van Engeland, & Buitelaar, 2006), 2) using a specific referral protocol, and 3) building a multidisciplinary diagnostic team. The programme was evaluated in a controlled study involving children in two regions (N = 2793, range 0-11 years). The main outcome variables were a difference in mean age at ASD diagnosis and a difference in the proportion of children diagnosed before 36 months. RESULTS: ASD was diagnosed 21 months (95% CI 9.6, 32.4) earlier in the experimental region than in the control region during the follow-up period, with the mean age at ASD diagnosis decreasing by 19.5 months (95% CI 10.5, 28.5) from baseline in the experimental region. Children from the experimental region were 9.4 times (95% CI 2.1, 41.3) more likely than children from the control region to be diagnosed before age 36 months after correction for baseline measurements. Most of these early diagnosed children had narrowly defined autism with mental retardation. CONCLUSIONS: The integrated early detection programme appears to be clinically relevant and led to the earlier detection of ASD, mainly in children with a low IQ.

Self-initiations in young children with autism during Pivotal Response Treatment with and without robot assistance.

LAY ABSTRACT: The initiation of social interaction is often defined as a core deficit of autism spectrum disorder. Optimizing these self-initiations is therefore a key component of Pivotal Response Treatment, an established intervention for children with autism spectrum disorder. However, little is known about the development of self-initiations during intervention and whether this development can be facilitated by robot assistance within Pivotal Response Treatment. The aim of this study was to (1) investigate the effect of Pivotal Response Treatment and robot-assisted Pivotal Response Treatment on self-initiations (functional and social) of young children with autism spectrum disorder over the course of intervention and (2) explore the relation between development in self-initiations and additional gains in general social-communicative skills. Forty-four children with autism spectrum disorder (aged 3-8 years) were included in this study. Self-initiations were assessed during parent-child interaction videos of therapy sessions and coded by raters who did not know which treatment (Pivotal Response Treatment or robot-assisted Pivotal Response Treatment) the child received. General social-communicative skills were assessed before start of the treatment, after 10 and 20 weeks of intervention and 3 months after the treatment was finalized. Results showed that self-initiations increased in both treatment groups, with the largest improvements in functional self-initiations in the group that received robot-assisted Pivotal Response Treatment. Increased self-initiations were related to higher parent-rated social awareness 3 months after finalizing the treatment.

Adherence and acceptability of a robot-assisted Pivotal Response Treatment protocol for children with autism spectrum disorder.

The aim of this study is to present a robot-assisted therapy protocol for children with ASD based on the current state-of-the-art in both ASD intervention research and robotics research, and critically evaluate its adherence and acceptability based on child as well as parent ratings. The robot-assisted therapy was designed based on motivational components of Pivotal Response Treatment (PRT), a highly promising and feasible intervention focused at training "pivotal" (key) areas such as motivation for social interaction and self-initiations, with the goal of establishing collateral gains in untargeted areas of functioning and development, affected by autism spectrum disorders. Overall, children (3-8 y) could adhere to the robot-assisted therapy protocol (Mean percentage of treatment adherence 85.5%), showed positive affect ratings after therapy sessions (positive in 86.6% of sessions) and high robot likability scores (high in 79.4% of sessions). Positive likability ratings were mainly given by school-aged children (H(1) = 7.91, p = .005) and related to the movements, speech and game scenarios of the robot. Parent ratings on the added value of the robot were mainly positive (Mean of 84.8 on 0-100 scale), while lower parent ratings were related to inflexibility of robot behaviour.

Social-communicative and attention problems in infancy and toddlerhood as precursors of preschool autistic traits.

This longitudinal study focused on early behavioural problems and autistic traits. In a stratified, population-derived sample of 119 children, mothers reported through questionnaires on externalizing, internalizing, and social-communicative characteristics of their child in infancy (14 months) and toddlerhood (37 months), and on autistic traits at preschool age (4-5 years). Children with consistently normal behaviour from infancy to toddlerhood showed lower autistic traits at preschool age than children with deviant behaviour on one or both time points. High autistic traits at preschool age were predominantly preceded by problems in interaction, communication, language, play, and affect in infancy and/or toddlerhood, but also by inattention in toddlerhood. Adequate support and specific interventions in these domains are needed in an attempt to diminish further derailment of the child's behaviour and development, and to prevent the full manifestation of ASD or related disorders such as ADHD.

[Early referral of toddlers on suspicion of autism spectrum disorder]. / Vroege verwijzing van peuters bij vermoeden van een autismespectrumstoornis.

Early signs of autism spectrum disorders (ASS) can become apparent at the baby and toddler stage and a diagnosis of ASS can already be made at this age. In practice, however, it often takes many years before children with ASS are diagnosed and as a result these children miss the benefits of early intervention. Reticence in referral and diagnosis seems to arise from personal factors, diagnostic problems and organisational factors. Here we discuss these factors and stress the importance of referral of very young children on suspicion of ASS to paediatric and paediatric psychiatric institutions with expertise in diagnosing and treating young children.

Sustainability of an early detection program for autism spectrum disorder over the course of 8 years.

The importance of early detection of autism spectrum disorder followed by early intervention is increasingly recognized. This quasi-experimental study evaluated the long-term effects of a program for the early detection of autism spectrum disorder (consisting of training of professionals and use of a referral protocol and screening instrument), to determine whether the positive effects on the age at referral were sustained after the program ended while controlling for overall changes in the number of referrals. Before, during, and after the program, the proportion of children referred before 3 years (versus 3-6 years) of age was calculated for children subsequently diagnosed with autism spectrum disorder ( N = 513) or another, non-autism spectrum disorder, condition ( N = 722). The odds of being referred before 3 years of age was higher in children with autism spectrum disorder than in children with another condition during the program than before (3.1, 95% confidence interval: 1.2-7.6) or after (1.7, 95% confidence interval: 1.0-3.0) the program but was not different before versus after the program. Thus, although the program led to earlier referral of children with autism spectrum disorder, after correction for other referrals, the effect was not sustained after the program ended. This study highlights the importance of continued investment in the early detection of autism spectrum disorder.

Does the Brief Observation of Social Communication Change help moving forward in measuring change in early autism intervention studies?

The field of early autism research is in dire need of outcome measures that adequately reflect subtle changes in core autistic behaviors. This article compares the ability of a newly developed measure, the Brief Observation of Social Communication Change (BOSCC), and the Autism Diagnostic Observation Schedule (ADOS) to detect changes in core symptoms of autism in 44 toddlers. The results provide encouraging evidence for the Brief Observation of Social Communication Change as a candidate outcome measure, as reflected in sufficient inter- and intra-rater reliability, independency from other child characteristics, and sensitivity to capture change. Although the Brief Observation of Social Communication Change did not evidently outperform the Autism Diagnostic Observation Schedule on any of these quality criteria, the instrument may be better able to capture subtle, individual changes in core autistic symptoms. The promising findings warrant further study of this new instrument.

Variation in the Early Trajectories of Autism Symptoms Is Related to the Development of Language, Cognition, and Behavior Problems.

OBJECTIVE: The objectives of this study were to model more homogeneous subgroups within autism spectrum disorder (ASD) based on early trajectories of core symptoms; and to further characterize these subgroups in terms of trajectories of language, cognition, co-occurring (attention-deficit/hyperactivity disorder [ADHD]-related) traits and clinical outcome diagnosis. METHOD: Children (N = 203) referred for possible ASD at ages 1 to 4 years were assessed at three time points at intervals ranging from 9 months to 3 years. Assessments included standardized measures for ASD (Autism Diagnostic Observation Schedule [ADOS]), language (ADOS-language item), nonverbal IQ (NV-IQ; different tests adequate to chronological/mental age), and parent-reported behavioral problems (Infant-Toddler Social and Emotional Assessment, Child Behavior Checklist). RESULTS: Latent-class growth curve analysis with ADOS total scores led to the identification of three main stable and two small improving groups: a severe-stable group (19.5% of sample)-the only group without considerable language improvement-showed persistent low NV-IQ and marked increase in attention problems over time; a moderate-stable group (21.7%) with below-average increasing NV-IQ; and a mild-stable group (48%) with stable-average NV-IQ and the highest scores on ADHD-related traits, whose ASD outcome diagnoses increased despite stable-low ASD scores. Two groups (each 5.4%) improved: one moved from severe to moderate ASD scores, and the other moved from moderate to mild/nonspectrum scores. Both of these groups improved on language, NV-IQ, and ADHD-related traits. CONCLUSION: Results support the high stability of ASD symptoms into various severity levels, but also highlight the significant contribution of non-ASD domains in defining and explaining the different ASD trajectories.

Autism Diagnostic Interview-Revised (ADI-R) Algorithms for Toddlers and Young Preschoolers: Application in a Non-US Sample of 1,104 Children.

The current study aimed to investigate the Autism Diagnostic Interview-Revised (ADI-R) algorithms for toddlers and young preschoolers (Kim and Lord, J Autism Dev Disord 42(1):82-93, 2012) in a non-US sample from ten sites in nine countries (n = 1,104). The construct validity indicated a good fit of the algorithms. The diagnostic validity was lower, with satisfactorily high specificities but moderate sensitivities. Young children with clinical ASD and lower language ability were largely in the mild-to-moderate or moderate-to-severe concern ranges of the ADI-R, nearly half of the older and phrase speech ASD-group fell into the little-to-no concern range. Although broadly the findings support the toddler algorithms, further work is required to understand why they might have different properties in different samples to further inform research and clinical use.

Gender and age differences in the core triad of impairments in autism spectrum disorders: a systematic review and meta-analysis.

Autism is an extensively studied disorder in which the gender disparity in prevalence has received much attention. In contrast, only a few studies examine gender differences in symptomatology. This systematic review and meta-analysis of 22 peer reviewed original publications examines gender differences in the core triad of impairments in autism. Gender differences were transformed and concatenated using standardized mean differences, and analyses were stratified in five age categories (toddlerhood, preschool children, childhood, adolescence, young adulthood). Boys showed more repetitive and stereotyped behavior as from the age of six, but not below the age of six. Males and females did not differ in the domain of social behavior and communication. There is an underrepresentation of females with ASD an average to high intelligence. Females could present another autistic phenotype than males. As ASD is now defined according to the male phenotype this could imply that there is an ascertainment bias. More research is needed into the female phenotype of ASD with development of appropriate instruments to detect and ascertain them.

Narrowly versus broadly defined autism spectrum disorders: differences in pre- and perinatal risk factors.

This study examined the differential contribution of pre- and perinatal risks in narrowly versus broadly defined autism spectrum disorder (ASD) and across core symptom domains, IQ and co-morbid problems. Children with a DSM-IV diagnosis of autistic disorder (AD) (n = 121) or pervasive developmental disorder not otherwise specified (PDD-NOS) (n = 75) were compared to a typical control sample (n = 311). Diagnoses were based on extensive assessments between 12 and 49 months of age (M = 33.3, SD = 6.4) and re-evaluated at 43-98 months (M = 68.1, SD = 10.7) in 70% of the cases. Compared with controls, cases with ASD were more likely to be firstborn and show a suboptimal condition after birth. Case mothers reported more infections and more stress during pregnancy. Although the ASD subgroups showed mostly overlapping risks, cases with PDD-NOS differed from those with AD by higher exposure to smoking during pregnancy (SDP) and by a negative association of smoking with IQ, regardless of confounders. SDP appears to contribute more to broadly defined (PDD-NOS) than to narrowly defined ASD (AD). Findings suggest differences in etiological contributors between ASD phenotypes.

How to use the ADI-R for classifying autism spectrum disorders? Psychometric properties of criteria from the literature in 1,204 Dutch children.

The algorithm of the Autism Diagnostic Interview-Revised provides criteria for autism versus non-autism according to DSM-IV. Criteria for the broader autism spectrum disorders are needed. This study investigated the validity of seven sets of criteria from the literature, in 1,204 Dutch children (aged 3-18 years) with and without mental retardation. The original criteria (Rutter et al. in ADI-R Autism Diagnostic Interview Revised. Manual. Western Psychological Services, Los Angeles, 2003) well discriminated ASD from non-ASD in MR. All other criteria (IMGSAC in Am Soc Hum Genet 69:570-581 2001; Sung et al. in Am J Hum Genet 76: 68-81, 2005; Risi et al. in J Am Acad Child Adolesc Psychiatry 45: 1094-1103, 2006) were sensitive at the cost of specificity, bearing the risk of overinclusiveness. In the group without MR, clinicians should decide whether sensitivity or specificity is aimed for, to choose the appropriate criteria. Including the Autism Diagnostic Observation Schedule revised algorithms in the classification, the specificity increases, at the cost of sensitivity. This study adds to a more valid judgment on which criteria to use for specific objectives.

A pilot study of abnormal growth in autism spectrum disorders and other childhood psychiatric disorders.

The aims of the current study were to examine whether early growth abnormalities are (a) comparable in autism spectrum disorders (ASD) and other childhood psychiatric disorders, and (b) specific to the brain or generalized to the whole body. Head circumference, height, and weight were measured during the first 19 months of life in 129 children with ASD and 59 children with non-ASD psychiatric disorders. Both groups showed comparable abnormal patterns of growth compared to population norms, especially regarding height and head circumference in relation to height. Thus abnormal growth appears to be related to psychiatric disorders in general and is mainly expressed as an accelerated growth of height not matched by an increase in weight or head circumference.