Treatment mechanism of the WHO caregiver skills training intervention for autism delivered in community settings.

This study examined the mechanism of effect of the WHO Caregiver Skills Training (CST) through secondary analysis of a pilot RCT conducted in community settings. Participants were 86 caregivers (77% mothers) of children with ASD (78% male, mean age: 44.8 months) randomized to CST (n = 43) or treatment as usual (n = 43). The primary outcomes, measured at baseline (t1), immediately post-intervention (t2), and 3 months post-intervention (t3), were derived from the coding of caregiver-child free play interactions with the Brief Observation of Social-Communication Change (BOSCC) and the Joint Engagement Rating Inventory scale (JERI). At t3 positive treatment main effects had been observed for caregiver skills supportive of the interaction and for flow of the interaction (JERI), albeit only non-significant changes in the expected direction for child outcomes: autism phenotypic behaviors (BOSCC), joint engagement and availability to interact (JERI). This study tested the theory of change of CST, hypothesizing that the intervention would lead to an improvement on all child and dyad outcomes through an increase in the caregiver skills supportive of the interaction. Serial mediation analyses revealed that the effect of the intervention was significantly influenced by change in caregiver skills. Participation in the intervention led to notable increases in caregiver skills at t2 and t3, which subsequently contributed to improvements at t3 in flow of the interaction, autism phenotypic behavior, joint engagement, and availability to interact. We confirmed our a priori hypothesis showing that change in caregiver skills significantly mediated the effect of treatment on the dyad primary outcome, as well as on the other child outcomes that had shown non-significant changes in the expected direction. Implications for intervention design and policy making in the context of public health services are discussed.

Does Treatment Fidelity of the Early Start Denver Model Impact Skill Acquisition in Young Children with Autism?

There is increasing evidence supporting the effectiveness of the Early Start Denver Model (ESDM) for children on the autism spectrum. However, substantial variability in response to the ESDM has been reported across participants. We examined the plausible yet untested hypothesis that variations in the fidelity level of therapists delivering the intervention contribute to variability in children's response to the ESDM. Videotaped sessions (n = 40) of toddlers on the autism spectrum who received the ESDM from trained therapists were coded to obtain measures of therapist fidelity and children's learning in response to the therapists' instruction. Variations in overall fidelity, along with variations in most items included in the ESDM fidelity checklist, contributed to the children's learning response during the sessions.

Early developmental delay in Leigh syndrome spectrum disorders is associated with poor clinical prognosis.

BACKGROUND: Leigh spectrum syndrome (LSS) is a primary mitochondrial disorder characterized by neurodevelopmental regression and metabolic stroke typically in early life. Developmental delay (DD) is known to follow episodes of neurologic regression in LSS, although primary developmental delay (pDD) has been rarely reported. We hypothesized that pDD precedes regression in a broader subset of LSS individuals and may associate with worse long-term educational outcomes. METHODS: From a retrospective cohort, subjects with pathogenic variant(s) in a nuclear or mitochondrial gene associated with LSS and consistent clinical manifestations and neuroradiological findings. Detailed developmental histories and neurologic outcomes were extracted. RESULTS: Of 69 LSS subjects, 47 (68.1%) had a history of pDD and 53 (76.8%) had neurodevelopmental regression. We identified 3 distinct developmental phenotypes: [1] pDD followed by regression (N = 31/69, 44.9%), [2] pDD without subsequent regression (16/69, 23.2%), [3] regression without pDD (N = 22/69, 31.9%). A history of pDD was associated with earlier disease onset (p = 0.0003) and worse educational outcomes (OR 22.14). CONCLUSION: LSS is associated with multiple developmental phenotypes and pDD is associated with negative educational outcomes. pDD occurring prior to neurologic regression suggests that mitochondrial energetics impact developmental trajectories prior to acute metabolic failure and regression, providing an opportunity for earlier diagnosis and/or therapeutic intervention.

The impact of using digitally-mediated social stories on the perceived competence and attitudes of parents and practitioners supporting children with autism.

A Social Story (SS) is a highly acceptable and widely used intervention by the autism community. Yet, inconsistent implementation of the intervention is reported to be one of the causes of variability in terms of outcome research, particularly in a naturalistic context. This study aimed to (1) investigate whether digitally-mediated social stories (SSs) can improve competence in developing and delivering a SS and thus contribute towards improved implementation, and (2) investigate the impact of the digitally-mediated SS on attitudes towards the SS intervention. Ninety-three participants took an initial pre-engagement survey. Forty-eight of these participants also complete a post-engagement survey. A pre-post design was utilised with the participants who completed both surveys. These 48 participants were invited to develop a digitally-mediated SS with the aim of exploring how digitally-mediated SSs impacted perceived competence and attitudes. Post-engagement data was collected two weeks after the pre-engagement data. Outcomes of this study indicate that both perceived competence and attitudes improved after engaging with digitally-mediated SSs. It is concluded that digitally-mediated SS not only impacted the integrity of how the intervention was delivered but also the beliefs in the participants' capabilities to develop and deliver a SS. Digitally-mediated SS, thus, has the potential to effectively support development and delivery whilst also addressing challenges related to intervention implementation in a naturalistic context.

Digitally-Mediated Social Stories Support Children on the Autism Spectrum Adapting to a Change in a 'Real-World' Context.

Social Stories™ (SS) is a widely used intervention for children on the autism spectrum. A preliminary survey of 103 practitioners highlighted that SS are often used to support adapting to a change. This study investigated the use of digitally-mediated SS to support ten children on the autism spectrum attending a school summer camp. Teacher perceptions of anxiety, understanding and closeness to the goal of the SS were assessed before and after the intervention (prior to the event). The pre- post-intervention comparisons highlighted significant improvements in child understanding, anxiety, and closeness to goal with medium-large effect sizes. The child's understanding and closeness to SS goal post-intervention related to their difficulties with the SS goal and their anxiety during the event.

[Pervasive refusal syndrome in children]. / Le syndrome de refus global de l'enfant.

Pervasive refusal syndrome is a rare clinical disorder affecting children and teenagers. It is characterised by social withdrawal and opposition which significantly affects how patients function and their treatment. The twenty or so cases reported in literature help to specify the main diagnostic and therapeutic elements. Early recognition of pervasive refusal syndrome is essential in order to treat these young patients as effectively as possible.

Systematic review of factors that may influence the outcomes and generalizability of parent-mediated interventions for young children with autism spectrum disorder.

Parent mediated interventions have the potential to positively influence the interactions and developmental outcomes of children with autism spectrum disorder (ASD). However, a range of factors relating to children, parents and caregivers, and study design may impact on outcomes and thus the generalizability of these interventions to the broader community. The objective of this review was to examine factors that may influence the feasibility, appropriateness, effectiveness, and generalizability of parent mediated interventions for children with ASD. We conducted a systematic review, yielding 41 articles. There was substantial variability in the intervention type, intensity, and study quality. Notably, 46 different inclusion/exclusion criteria were reported across studies including factors relating to children's development, access to other services, comorbidities, parental factors, and access to the intervention. Fifteen articles included examination of 45 different factors potentially associated with, or influencing, intervention outcomes including child (e.g., language skills, ASD severity, cognition) and parent (e.g., adherence and fidelity, education) factors. Although there is clear evidence for an increasingly sophisticated (e.g., systematic phased research for some interventions) and diverse (e.g., studies in geographical diverse contexts including low-resource communities) approach to research examining parent mediated interventions, there remains a need for improved study quality and measurement consistency in research, including a detailed examination of factors that may predict, moderate, and mediate intervention effectiveness for children and their parents. Autism Res 2019, 12: 1304-1321. © 2019 International Society for Autism Research, Wiley Periodicals, Inc. LAY SUMMARY: Parent mediated interventions-in which parents adapt their own behavior or deliver interventions to help their children learn-appear to be effective for some children with autism spectrum disorder. In this review, we identified a range of child, parent, and study design factors that may influence intervention outcomes and ultimately the uptake of these approaches in the community. We suggest that research in this area could be further improved by ensuring that studies include diverse groups of children and parents, and by using study designs that help to establish not only if interventions work, but for whom they work best and why.

Tramas de la constitución subjetiva y la complejidad del diagnóstico de niños desde un enfoque relacional (parte 2) / Networks of subjetive constitution and the complexity of the diagnosis of children from a relational perspective (2nd part)

Este trabajo amplia uno precedente. Aporta al abordaje del diagnóstico y sus enlaces con las vicisitudes de la recomendación terapéutica; en términos de diferenciar entre dificultades por déficits, conflictos o reconstrucciones subjetivas en curso. Para trazar ejes conceptuales que delinean estas diferencias, como figura didáctica, puntualizamos observables evolutivos, organizados en torno a las primeras ligazones amorosas, un segundo tiempo de diferenciaciones amorosas y un tercero de descubrimiento de la alteridad del otro. Observables que se constituyen las capas profundas de la mente, coexisten y transforman a lo largo de la vida. En pos de señalar la importancia de elaborar diagnósticos que den cuenta de lo constituido y sus transformaciones, de déficits y/o dificultades nos focalizamos en la niñez. Dos supuestos básicos guían esta presentación; en la vida de una persona el diagnóstico nunca resulta neutro, es terapéutico o iatrogénico, tanto desarrollo como trauma devienen en la dimensión relacional intersubjetiva

This work expands one precedent. It contributes to the approach of the diagnosis and its links with the vicissitudes of the therapeutic recommendation; In terms of differentiating between difficulties due to ongoing deficits, conflicts or subjective reconstructions. As a didactic device, we decided to consider developmental aspects observable across childhood in order to outline core concepts that allow us to improve our analysis of these aspects. Said observable developmental aspects relate to the first affectionate bonds, a second stage of affectionate differentiations and a third stage of the discovery of otherness. These dimensions are pointed out as emergent in the frame of complex networks of nonlinear subjective constitution processes. These observable developmental aspects exist in the deep layers of our minds, coexist and transform along life. In this work, our descriptions focus on the first two dimensions. Our purpose is to highlight the importance of making diagnoses that account for what is established and its transformations, deficiencies and/or difficulties. Two basic assumptions guide this presentation: that in a person’s life, a diagnosis is never neutral; it is either therapeutic or iatrogenic, and that both development and trauma occur in an intersubjective relational dimension

Early intensive behavioral intervention (EIBI) for young children with autism spectrum disorders (ASD).

BACKGROUND: The rising prevalence of autism spectrum disorders (ASD) increases the need for evidence-based behavioral treatments to lessen the impact of symptoms on children's functioning. At present, there are no curative or psychopharmacological therapies to effectively treat all symptoms of the disorders. Early intensive behavioral intervention (EIBI) is a treatment based on the principles of applied behavior analysis. Delivered for multiple years at an intensity of 20 to 40 hours per week, it is one of the more well-established treatments for ASD. This is an update of a Cochrane review last published in 2012. OBJECTIVES: To systematically review the evidence for the effectiveness of EIBI in increasing functional behaviors and skills, decreasing autism severity, and improving intelligence and communication skills for young children with ASD. SEARCH METHODS: We searched CENTRAL, MEDLINE, Embase, 12 additional electronic databases and two trials registers in August 2017. We also checked references and contacted study authors to identify additional studies. SELECTION CRITERIA: Randomized control trials (RCTs), quasi-RCTs, and controlled clinical trials (CCTs) in which EIBI was compared to a no-treatment or treatment-as-usual control condition. Participants must have been less than six years of age at treatment onset and assigned to their study condition prior to commencing treatment. DATA COLLECTION AND ANALYSIS: We used standard methodological procedures expected by Cochrane.We synthesized the results of the five studies using a random-effects model of meta-analysis, with a mean difference (MD) effect size for outcomes assessed on identical scales, and a standardized mean difference (SMD) effect size (Hedges' g) with small sample correction for outcomes measured on different scales. We rated the quality of the evidence using the GRADE approach. MAIN RESULTS: We included five studies (one RCT and four CCTs) with a total of 219 children: 116 children in the EIBI groups and 103 children in the generic, special education services groups. The age of the children ranged between 30.2 months and 42.5 months. Three of the five studies were conducted in the USA and two in the UK, with a treatment duration of 24 months to 36 months. All studies used a treatment-as-usual comparison group.Primary outcomesThere is low quality-evidence at post-treatment that EIBI improves adaptive behaviour (MD 9.58 (assessed using Vineland Adaptive Behavior Scale (VABS) Composite; normative mean = 100, normative SD = 15), 95% confidence interval (CI) 5.57 to 13.60, P < 0.0001; 5 studies, 202 participants), and reduces autism symptom severity (SMD -0.34, 95% CI -0.79 to 0.11, P = 0.14; 2 studies, 81 participants; lower values indicate positive effects) compared to treatment as usual.No adverse effects were reported across studies.Secondary outcomesThere is low-quality evidence at post-treatment that EIBI improves IQ (MD 15.44 (assessed using standardized IQ tests; scale 0 to 100, normative SD = 15), 95% CI 9.29 to 21.59, P < 0.001; 5 studies, 202 participants); expressive (SMD 0.51, 95% CI 0.12 to 0.90, P = 0.01; 4 studies, 165 participants) and receptive (SMD 0.55, 95% CI 0.23 to 0.87, P = 0.001; 4 studies, 164 participants) language skills; and problem behaviour (SMD -0.58, 95% CI -1.24 to 0.07, P = 0.08; 2 studies, 67 participants) compared to treatment as usual. AUTHORS' CONCLUSIONS: There is weak evidence that EIBI may be an effective behavioral treatment for some children with ASD; the strength of the evidence in this review is limited because it mostly comes from small studies that are not of the optimum design. Due to the inclusion of non-randomized studies, there is a high risk of bias and we rated the overall quality of evidence as 'low' or 'very low' using the GRADE system, meaning further research is very likely to have an important impact on our confidence in the estimate of effect and is likely to change the estimate.It is important that providers of EIBI are aware of the current evidence and use clinical decision-making guidelines, such as seeking the family's input and drawing upon prior clinical experience, when making recommendations to clients on the use EIBI. Additional studies using rigorous research designs are needed to make stronger conclusions about the effects of EIBI for children with ASD.

Process Evaluation of the BOOST-A™ Transition Planning Program for Adolescents on the Autism Spectrum: A Strengths-Based Approach.

A process evaluation was conducted to determine the effectiveness, usability, and barriers and facilitators related to the Better OutcOmes & Successful Transitions for Autism (BOOST-A™), an online transition planning program. Adolescents on the autism spectrum (n = 33) and their parents (n = 39) provided feedback via an online questionnaire. Of these, 13 participants were interviewed to gain in-depth information about their experiences. Data were analyzed using descriptive statistics and thematic analysis. Four themes were identified: (i) taking action to overcome inertia, (ii) new insights that led to clear plans for the future, (iii) adolescent empowerment through strengths focus, and (iv) having a champion to guide the way. The process evaluation revealed why BOOST-A™ was beneficial to some participants more than others. Trial registration #ACTRN12615000119594.

Virtual Environment for Social Information Processing: Assessment of Children with and without Autism Spectrum Disorders.

Social information processing (SIP) skills are critical for developing and maintaining peer relationships. Building on existing assessment techniques, Virtual Environment for SIP (VESIPTM ), a simulation-based assessment that immerses children in social decision-making scenarios, was developed. This study presents preliminary evidence of VESIP's usefulness for measuring SIP skills in children with and without autism spectrum disorders (ASD). Twenty-one children with ASD and 29 control children participated. It was hypothesized that (a) children (8-12 years old), with and without ASD, would understand and interact effectively with VESIP; (b) VESIP scores would be reliable in both populations; and (c) children with ASD would score lower on SIP domains than typically developing peers. Results supported these hypotheses. Finally, response bias was also evaluated, showing that children with ASD have different problem-solving strategies than their peers. VESIP has great potential as a scalable assessment of SIP strengths and challenges in children with and without ASD. Autism Res 2018, 11: 305-317. © 2017 International Society for Autism Research, Wiley Periodicals, Inc. LAY SUMMARY: Children with autism spectrum disorders (ASDs) often struggle interpreting and responding to social situations. The present study suggests that an animated, simulation-based assessment approach is an effective way to measure how children with or without ASDs problem-solve challenging social situations. VESIP is an easy-to-use assessment tool that can help practitioners understand a child's particular strengths and weaknesses.

Symbolic Play in School-Aged Minimally Verbal Children with Autism Spectrum Disorder.

Few interventions exist for school-aged minimally verbal children with autism spectrum disorder (ASD). Even though play skills are associated with children's production of language, few studies have focused on play for minimally verbal children. Fifty-eight minimally verbal children with ASD received a naturalistic developmental behavioral intervention. Children were randomized to receive a speech generating device in the context of the intervention or not. Children in both conditions improved in play skills at exit. Children demonstrated an increase in play skills in proximal (sessions) and distal (during blind assessment) contexts. Minimally verbal children with ASD can improve their play skills within a targeted intervention. Increases in symbolic play were associated with increases in expressive language skills.

Una experiencia grupal en pacientes con trastornos generalizados del desarrollo / A group experience with patients with pervasive developmental disorders / Una experiència grupal en pacients amb trastorns generalitzats del desenvolupament

Los trastornos generalizados del desarrollo presentan dificultades relacionales y de simbolización (Alcácer y Viloca, 2014), que se agravan con los cambios físicos de la pubertad. Nos planteamos un abordaje grupal centrado en la relación a través de una tarea que favorezca el proceso de simbolización. Observamos en los pacientes una mejoría en la capacidad de interesarse por el otro, una menor necesidad de contención con psicofármacos, un aumento de la capacidad de abstracción y una mayor simbolización. La hipótesis consiste en que la observación de imágenes y discusión sobre las mismas en grupo puede ser un elemento facilitador del trabajo del mundo interno y la relación entre los integrantes del grupo

Pervasive Developmental Disorders present relational and symbolic difficulties (Alcácer and Viloca, 2014), which are aggravated by the physical changes of puberty. We propose a relationship-centered group approach employing a task that favors the process of symbolization. We observed improvements in the patients in their capacity to be interested in others, a decreased need for containment with psychoactive drugs, an increase in the capacity for abstract thought and greater symbolization. The hypothesis is that the observation of images and discussion about them in a group might be an element that facilitates the work of the internal world and the relations between the members of the group

Els trastorns generalitzats del desenvolupament presenten dificultats relacionals I de simbolització (Alcácer I Viloca, 2014), que s'agreugen amb els canvis físics de la pubertat. Els plantegem un abordatge grupal centrat en la relació mitjançant una tasca que afavoreixi el procés de simbolització. Observem en els pacients una millora en la capacitat d'interessar-se per l'altre, una menor necessitat de contenció amb psicofàrmacs, un augment de la capacitat d'abstracció I una major simbolització. La hipòtesi consisteix en el fet que l'observació d'imatges I la discussió sobre aquestes en grup pot ser un element facilitador del treball del món intern I la relació entre els integrants del grup

[Left temporal arachnoid cyst and specific learning disorders associated with Pervasive Developmental Disorders - Not Otherwise Specified (PDD-NOS): contributions of an integrative neuropsychomotor, neuropsychological, psychopathological and neurosurgical approach about a case report in a child (François)]. / Kyste arachnoïdien temporal gauche et troubles spécifiques des apprentissages associés à un trouble envahissant du développement non spécifié (TED-NoS) : apports d'une approche intégrative neuro-psychomotrice neuropsychologique, psychopathologique et neurochirurgicale à propos d'une observation chez un enfant (le cas François).

Left temporal arachnoid cyst and specific learning disorders associated with pervasive developmental disorders - not otherwise specified (PDD-NOS): contributions of an integrative neuro-psychomotor, neuropsychological, psychopathological and neurosurgical approach about a case report in a child (François). With DSM-IV and DSM-IV-TR, the terminology of pervasive developmental disorders (PDD) covers two main categories of infantile disorders: disorders of "strictly" autistic nature and pervasive developmental disorders - not otherwise specified (PDD-NOS). Under the terminology of multiple complex developmental disorder (MCDD), it is proposed to classify children presenting symptoms approaching the psychotic disharmonies and usually diagnosed as PDD-NOS. Such a category of developmental disorders is now included without nosographic distinction in the autistic spectrum in the Diagnostic and Statistical Manual of mental disorders (DSM-V). CASE REPORT: We are reporting a case report of a 6-year-old boy which shows a PDD-NoS/MCDD complex symptomatology type. This child presents multiple disorders: minor neurological signs (soft signs), neuro-psychomotor disorders, developmental coordination disorder (DCD), communication, thought, and regulation of emotions disorders, attention deficit disorders (ADD); in the presence of a high verbal intellectual potential, which makes it difficult to establish a clear diagnosis. A cerebral magnetic resonance imaging (MRI) was carried out due to the presence of minor neurological signs (soft signs) and of neurodevelopmental multiple disorders. The MRI revealed a voluminous arachnoid temporo-polar left cyst with a marked mass effect on the left temporal lobe. DISCUSSION: A neurosurgical intervention allowed to observe the gradual disappearance of the specific symptomatology (in particular soft signs, neuro-psychomotor functions and autistic symptoms) secondary to the interference of the cyst's pressure with intracranial areas involving neurological and psychopathological abnormalities, underlying at the same time the reversibility of the disorders after decompression as demonstrated in some studies. There are always, with a quantitative and qualitative decrease, an emotional dysregulation, a DCD, an ADD as well as impairments in the executive functions. CONCLUSION: This clinical case underlines the necessity of an evaluation in a transdisciplinary way and to follow the developmental evolution of the child in order to focus adapted therapeutics. Furthermore, with neurodevelopmental disorders not specified, it is important to examine the presence of soft signs with standardized neuro-psychomotor assessment, and then, to propose an MRI investigation. To our knowledge, this is the first report in the literature with a school age child of an unusual association between a temporal arachnoid cyst associated with PDD-NOS/MCDD.

Advancing the understanding of autism disease mechanisms through genetics.

Progress in understanding the genetic etiology of autism spectrum disorders (ASD) has fueled remarkable advances in our understanding of its potential neurobiological mechanisms. Yet, at the same time, these findings highlight extraordinary causal diversity and complexity at many levels ranging from molecules to circuits and emphasize the gaps in our current knowledge. Here we review current understanding of the genetic architecture of ASD and integrate genetic evidence, neuropathology and studies in model systems with how they inform mechanistic models of ASD pathophysiology. Despite the challenges, these advances provide a solid foundation for the development of rational, targeted molecular therapies.

Brief Report: Joint Attention and Information Processing in Children with Higher Functioning Autism Spectrum Disorders.

Theory suggests that information processing during joint attention may be atypical in children with Autism Spectrum Disorder (ASD). This hypothesis was tested in a study of school-aged children with higher functioning ASD and groups of children with symptoms of ADHD or typical development. The results indicated that the control groups displayed significantly better recognition memory for pictures studied in an initiating joint attention (IJA) rather than responding to joint attention (RJA) condition. This effect was not evident in the ASD group. The ASD group also recognized fewer pictures from the IJA condition than controls, but not the RJA condition. Atypical information processing may be a marker of the continued effects of joint attention disturbance in school aged children with ASD.

Longitudinal Effects of Adaptive Interventions With a Speech-Generating Device in Minimally Verbal Children With ASD.

There are limited data on the effects of adaptive social communication interventions with a speech-generating device in autism. This study is the first to compare growth in communications outcomes among three adaptive interventions in school-age children with autism spectrum disorder (ASD) who are minimally verbal. Sixty-one children, ages 5-8 years, participated in a sequential, multiple-assignment randomized trial (SMART). All children received a developmental behavioral communication intervention: joint attention, symbolic play, engagement and regulation (JASP) with enhanced milieu teaching (EMT). The SMART included three 2-stage, 24-week adaptive interventions with different provisions of a speech-generating device (SGD) in the context of JASP+EMT. The first adaptive intervention, with no SGD, initially assigned JASP+EMT alone, then intensified JASP+EMT for slow responders. In the second adaptive intervention, slow responders to JASP+EMT were assigned JASP+EMT+SGD. The third adaptive intervention initially assigned JASP+EMT+SGD; then intensified JASP+EMT+SGD for slow responders. Analyses examined between-group differences in change in outcomes from baseline to Week 36. Verbal outcomes included spontaneous communicative utterances and novel words. Nonlinguistic communication outcomes included initiating joint attention and behavior regulation, and play. The adaptive intervention beginning with JASP+EMT+SGD was estimated as superior. There were significant (p < .05) between-group differences in change in spontaneous communicative utterances and initiating joint attention. School-age children with ASD who are minimally verbal make significant gains in communication outcomes with an adaptive intervention beginning with JASP+EMT+SGD. Future research should explore mediators and moderators of the adaptive intervention effects and second-stage intervention options that further capitalize on early gains in treatment.