1. Bangerter A, Manyakov NV, Lewin D, Boice M, Skalkin A, Jagannatha S, Chatterjee M, Dawson G, Goodwin MS, Hendren R, Leventhal B, Shic F, Ness S, Pandina G. {{Caregiver Daily Reporting of Symptoms in Autism Spectrum Disorder: Observational Study Using Web and Mobile Apps}}. {JMIR Ment Health};2019 (Mar 26);6(3):e11365.
BACKGROUND: Currently, no medications are approved to treat core symptoms of autism spectrum disorder (ASD). One barrier to ASD medication development is the lack of validated outcome measures able to detect symptom change. Current ASD interventions are often evaluated using retrospective caregiver reports that describe general clinical presentation but often require recall of specific behaviors weeks after they occur, potentially reducing accuracy of the ratings. My JAKE, a mobile and Web-based mobile health (mHealth) app that is part of the Janssen Autism Knowledge Engine-a dynamically updated clinical research system-was designed to help caregivers of individuals with ASD to continuously log symptoms, record treatments, and track progress, to mitigate difficulties associated with retrospective reporting. OBJECTIVE: My JAKE was deployed in an exploratory, noninterventional clinical trial to evaluate its utility and acceptability to monitor clinical outcomes in ASD. Hypotheses regarding relationships among daily tracking of symptoms, behavior, and retrospective caregiver reports were tested. METHODS: Caregivers of individuals with ASD aged 6 years to adults (N=144) used the My JAKE app to make daily reports on their child’s sleep quality, affect, and other self-selected specific behaviors across the 8- to 10-week observational study. The results were compared with commonly used paper-and-pencil scales acquired over a concurrent period at regular 4-week intervals. RESULTS: Caregiver reporting of behaviors in real time was successfully captured by My JAKE. On average, caregivers made reports 2-3 days per week across the study period. Caregivers were positive about their use of the system, with over 50% indicating that they would like to use My JAKE to track behavior outside of a clinical trial. More positive average daily reporting of overall type of day was correlated with 4 weekly reports of lower caregiver burden made at 4-week intervals (r=-0.27, P=.006, n=88) and with ASD symptoms (r=-0.42, P<.001, n=112). CONCLUSIONS: My JAKE reporting aligned with retrospective Web-based or paper-and-pencil scales. Use of mHealth apps, such as My JAKE, has the potential to increase the validity and accuracy of caregiver-reported outcomes and could be a useful way of identifying early changes in response to intervention. Such systems may also assist caregivers in tracking symptoms and behavior outside of a clinical trial, help with personalized goal setting, and monitoring of progress, which could collectively improve understanding of and quality of life for individuals with ASD and their families. TRIAL REGISTRATION: ClinicalTrials.gov NCT02668991; https://clinicaltrials.gov/ct2/show/NCT02668991. Lien vers le texte intégral (Open Access ou abonnement)
2. Crane L, Jones L, Prosser R, Taghrizi M, Pellicano E. {{Parents’ views and experiences of talking about autism with their children}}. {Autism};2019 (Mar 27):1362361319836257.
The way an autism diagnosis is disclosed to parents has been found to play a crucial role in their acceptance of, and the way they cope with, their child’s diagnosis. Yet, research into parents’ subsequent experiences of disclosing a diagnosis to their children, and talking to their families about autism more generally, is limited. Using an online survey, the current study examined 558 parents’ experiences of talking about autism with their autistic and non-autistic children. Results demonstrated that most parents ( n = 379, 67.9%) had told their autistic children about their diagnosis. Despite few parents ( n = 163, 20.4%) receiving advice or support regarding the disclosure of the diagnosis, those that had disclosed felt satisfied with the process ( n = 319, 84.2%) and felt confident in talking about autism with their children ( n = 339, 92.4%). Those who had not told their autistic children about the diagnosis largely planned to discuss this with their child in the future ( n = 100, 73.5%), felt confident in doing so ( n = 95, 70.9%) and were satisfied with their decision ( n = 95, 70.4%). Analysis of open-ended data, using thematic analysis, highlighted the importance of openness and the need to tailor explanations to individual children’s needs, while acknowledging that disclosure could often be challenging for parents.
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3. Dai YG, Burke JD, Naigles L, Eigsti IM, Fein DA. {{Language Abilities in Monolingual- and Bilingual- Exposed Children with Autism or Other Developmental Disorders}}. {Res Autism Spectr Disord};2018 (Nov);55:38-49.
Background: Parents and providers are sometimes concerned that exposure to two languages will impair language acquisition in children with autism spectrum disorder (ASD) or other developmental disorders (DD). However, research to date suggests that language milestones and abilities are unaffected by this exposure. The current study explored language abilities in toddlers with ASD or DD exposed to one versus multiple languages, prior to intervention. To our knowledge, this is the largest investigation of language learning in bilingual-exposed (BE) children with ASD. Methods: Participants were 388 children evaluated as part of a larger study on the early detection of ASD. Parents were asked to list all languages that primary caretakers use to communicate with their child. One hundred six BE children (57 ASD, 49 DD) were compared to 282 monolingual-exposed (ME) children (176 ASD, 106 DD). The Mullen Scales of Early Learning assessed nonverbal and verbal abilities. Multiple regression was used to evaluate the relationship of BE to language abilities, beyond the influence of nonverbal cognitive abilities, diagnosis, and socioeconomic status. Results: Results showed greater language impairment in ASD than DD, but no main effect for language exposure group nor any interaction of language group by diagnosis. Results remained consistent after controlling for socioeconomic status. Conclusion: This study suggests that bilingual caregivers can communicate with their children in both languages without adverse effects on their children’s language functioning.
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4. Danforth AL. {{Embracing Neurodiversity in Psychedelic Science: A Mixed-Methods Inquiry into the MDMA Experiences of Autistic Adults}}. {J Psychoactive Drugs};2019 (Mar 25):1-9.
This exploratory inquiry analyzed subjective experiences autistic adults reported after they took the drug 3,4-methylenedioxymethamphetamine (MDMA), also known as ecstasy, in nonclinical settings. Using a secure, globally available website, this study collected data from participants in 13 countries who were experienced with MDMA (n = 100). A subset of survey respondents (n = 24) were then invited to participate in qualitative interviews. The researcher applied thematic content analysis of interview transcripts to create a comprehensive account of emergent themes. MDMA has well-documented acute effects that promote pro-social attitudes such as caring and trust in neurotypical, or typically developing, populations. Findings from this study suggested that MDMA-assisted therapy may be an effective catalyst in autistic adults for intra- and interpersonal change. In addition, participants reported accounts of lasting transformation and healing from conditions such as trauma and social anxiety that are common in autistic populations. No participants reported long-term adverse outcomes as a result of using MDMA/ecstasy. Qualitative findings support a case for future clinical trials of MDMA-assisted therapy with autistic adults who present with social adaptability challenges.
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5. Das I, Estevez MA, Sarkar AA, Banerjee-Basu S. {{A multifaceted approach for analyzing complex phenotypic data in rodent models of autism}}. {Mol Autism};2019;10:11.
Autism (MIM 209850) is a multifactorial disorder with a broad clinical presentation. A number of high-confidence ASD risk genes are known; however, the contribution of non-genetic environmental factors towards ASD remains largely uncertain. Here, we present a bioinformatics resource of genetic and induced models of ASD developed using a shared annotation platform. Using this data, we depict the intricate trends in the research approaches to analyze rodent models of ASD. We identify the top 30 most frequently studied phenotypes extracted from rodent models of ASD based on 787 publications. As expected, many of these include animal model equivalents of the « core » phenotypes associated with ASD, such as impairments in social behavior and repetitive behavior, as well as several comorbid features of ASD including anxiety, seizures, and motor-control deficits. These phenotypes have also been studied in models based on a broad range of environmental inducers present in the database, of which gestational exposure to valproic acid (VPA) and maternal immune activation models comprising lipopolysaccharide (LPS) and poly I:C are the most studied. In our unique dataset of rescue models, we identify 24 pharmaceutical agents tested on established models derived from various ASD genes and CNV loci for their efficacy in mitigating symptoms relevant for ASD. As a case study, we analyze a large collection of Shank3 mouse models providing a high-resolution view of the in vivo role of this high-confidence ASD gene, which is the gateway towards understanding and dissecting the heterogeneous phenotypes seen in single-gene models of ASD. The trends described in this study could be useful for researchers to compare ASD models and to establish a complete profile for all relevant animal models in ASD research.
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6. Ecker C. {{Notice of Retraction and Replacement: Ecker et al. Association between the probability of autism spectrum disorder and normative sex-related phenotypic diversity in brain structure. JAMA Psychiatry. 2017;74(4):329-338}}. {JAMA Psychiatry};2019 (Mar 27)
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7. Glaze DG, Neul JL, Kaufmann WE, Berry-Kravis E, Condon S, Stoms G, Oosterholt S, Della Pasqua O, Glass L, Jones NE, Percy AK. {{Double-blind, randomized, placebo-controlled study of trofinetide in pediatric Rett syndrome}}. {Neurology};2019 (Mar 27)
OBJECTIVE: To determine safety, tolerability, and pharmacokinetics of trofinetide and evaluate its efficacy in female children/adolescents with Rett syndrome (RTT), a debilitating neurodevelopmental condition for which no pharmacotherapies directed at core features are available. METHODS: This was a phase 2, multicenter, double-blind, placebo-controlled, parallel-group study, in which safety/tolerability, pharmacokinetics, and clinical response to trofinetide were characterized in 82 children/adolescents with RTT, aged 5 to 15 years. Sixty-two participants were randomized 1:1:1:1 to receive placebo twice a day (bid) for 14 days, followed by placebo, 50, 100, or 200 mg/kg bid of trofinetide for 42 days. Following blinded safety data review, 20 additional participants were randomized 1:1 to the 200 mg/kg or placebo bid groups. Safety assessments included adverse events, clinical laboratory tests, physical examinations, and concomitant medications. Clinician- and caregiver-based efficacy measurements assessed clinically relevant, phenotypic dimensions of impairment of RTT. RESULTS: All dose levels were well tolerated and generally safe. Trofinetide at 200 mg/kg bid showed statistically significant and clinically relevant improvements relative to placebo on the Rett Syndrome Behaviour Questionnaire, RTT-Clinician Domain Specific Concerns-Visual Analog Scale, and Clinical Global Impression Scale-Improvement. Exploratory analyses suggested that observed changes correlated with trofinetide exposure. CONCLUSION: These results, together with those from a previous adolescent/adult trial, indicate trofinetide’s potential for treating core RTT symptoms and support further trials. CLASSIFICATION OF EVIDENCE: This study provides Class I evidence that for children/adolescents with RTT, trofinetide was safe, well-tolerated, and demonstrated improvement over placebo at 200 mg/kg bid in functionally important dimensions of RTT.
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8. Hilton CL, Ratcliff K, Collins DM, Flanagan J, Hong I. {{Flourishing in children with autism spectrum disorders}}. {Autism Res};2019 (Mar 26)
Flourishing is an indicator of positive mental health and is important for children’s development and well-being. We used variables from the National Survey of Children’s Health 2016 as indicators of flourishing (difficulty making friends, is bullied, bullies others, shares ideas with family, argues, finishes tasks, does all homework, shows curiosity, stays calm, and cares about doing well in school) to compare differences in parent perceptions of their children with and without autism spectrum disorder (ASD). We anticipate that these findings will help identify intervention targets to support the well-being of individuals with ASD. Children between 6 and 17 years of age, without intellectual disability, brain injury, cerebral palsy, or Down syndrome were included. Total participants were 34,171 controls (male/female = 17,116/17,155) and 812 with ASD (male/female = 668/144). Factor analysis resulted in three-factor structures (social competence, behavioral control, and school motivation) with good model fit (root mean square error of approximation = 0.08, comparative fit index = 0.92, Tucker-Lewis index = 0.89). The multivariate regression model and propensity score with inverse probability of treatment weighting (PS-IPTW) method revealed that children with ASD had lower scores in the social competence and behavioral control factors compared to the control group (all P < 0.05). However, no significant differences were found in the school motivation factor between the two groups (P > 0.05) in both multivariate regression model and PS-IPTW method. Findings suggest that social competence and behavioral control are indicators of flourishing and are important intervention targets to increase flourishing among children with ASD. Autism Res 2019, 00: 1-15. (c) 2019 International Society for Autism Research, Wiley Periodicals, Inc. LAY SUMMARY: Flourishing is an indicator of positive mental health and is important for children’s development and well-being. We used variables from The National Survey of Children’s Health 2016 to examine differences in parent perceptions of the indicators of flourishing (difficulty making friends, is bullied, bullies others, shares ideas with family, argues, finishes tasks, does all homework, shows curiosity, stays calm, and cares about doing well in school) between children with and without autism spectrum disorders (ASD). We anticipate that this information will help to identify therapeutic targets to support the well-being of individuals with ASD. Children between 6 and 17 years old, without intellectual disability (ID), brain injury (BI), cerebral palsy (CP), or Down syndrome (DS) were included. From the total (N = 50,212), we excluded children under age 6 (n = 14,494), those who once, but do not currently have ASD (n = 81), and those with ID (n = 432), BI (n = 170), CP (n = 35), and DS (n = 17), resulting in 34,983 records used. Total participants, age 6-17 years, were 34,171 controls (male/female = 17,116/17,155) and 812 with ASD (male/female = 668/144). Factor analysis resulted in the identification of three flourishing categories among the indicator variables (social competence, behavioral control, and school motivation). Children with ASD had lower scores in the social competence and behavioral control factors compared to the control group. However, there were no significant differences in the school motivation factor between the two groups. Findings suggest that social competence and behavioral control are indicators of flourishing and are important intervention targets to increase flourishing among children with ASD.
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9. Horvath G, Otrokocsi L, Beko K, Baranyi M, Kittel A, Fritz-Ruenes PA, Sperlagh B. {{P2X7 Receptors Drive Poly(I:C) Induced Autism-like Behavior in Mice}}. {J Neurosci};2019 (Mar 27);39(13):2542-2561.
Maternal immune activation (MIA) is a principal environmental risk factor contributing to autism spectrum disorder (ASD), which compromises fetal brain development at critical periods of pregnancy and might be causally linked to ASD symptoms. We report that endogenous activation of the purinergic ion channel P2X7 (P2rx7) is necessary and sufficient to transduce MIA to autistic phenotype in male offspring. MIA induced by poly(I:C) injections to P2rx7 WT mouse dams elicited an autism-like phenotype in their offspring, and these alterations were not observed in P2rx7-deficient mice, or following maternal treatment with a specific P2rx7 antagonist, JNJ47965567. Genetic deletion and pharmacological inhibition of maternal P2rx7s also counteracted the induction of IL-6 in the maternal plasma and fetal brain, and disrupted brain development, whereas postnatal P2rx7 inhibition alleviated behavioral and morphological alterations in the offspring. Administration of ATP to P2rx7 WT dams also evoked autistic phenotype, but not in KO dams, implying that P2rx7 activation by ATP is sufficient to induce autism-like features in offspring. Our results point to maternal and offspring P2rx7s as potential therapeutic targets for the early prevention and treatment of ASD.SIGNIFICANCE STATEMENT Autism spectrum disorder (ASD) is a neurodevelopmental psychiatric disorder caused by genetic and environmental factors. Recent studies highlighted the importance of perinatal risks, in particular, maternal immune activation (MIA), showing strong association with the later emergence of ASD in the affected children. MIA could be mimicked in animal models via injection of a nonpathogenic agent poly(I:C) during pregnancy. This is the first report showing the key role of a ligand gated ion channel, the purinergic P2X7 receptor in MIA-induced autism-like behavioral and biochemical features. We show that genetic or pharmacological inhibition of both maternal and offspring P2X7 receptors could reverse the compromised brain development and autistic phenotype pointing to new possibilities for prevention and treatment of ASD.
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10. Love AMA, Toland MD, Usher EL, Campbell JM, Spriggs AD. {{Can I teach students with Autism Spectrum Disorder?: Investigating teacher self-efficacy with an emerging population of students}}. {Res Dev Disabil};2019 (Mar 23);89:41-50.
Currently, 1 in 68 children in the U.S. is diagnosed with Autism Spectrum Disorder (ASD; Centers for Disease Control & Prevention, 2015) and this growing population of learners has been noted as one of the most challenging groups to teach. Teacher self-efficacy, the belief teachers hold about their instructional capabilities, has been shown to differ according to contextual factors, such as the type of students teachers face. The purpose of this investigation was to develop an instrument that can used to measure teachers’ self-efficacy for effectively working with students with ASD. Study 1 involved the development and evaluation of a new instrument, the Teacher Self-Efficacy for Students with Autism Scale (TSEAS) with a sample of general and special education teachers in the U.S. (N = 120). Study 2 involved a cross-validation of the measure with teachers in Australia (N = 85). Results indicated that the scale represented a unidimensional construct in both studies. Self-efficacy for teaching students with ASD was distinct from, though positively related to, general teaching self-efficacy, job satisfaction, and self-regulation. Using a student-specific teaching self-efficacy measure might provide more useful information for supporting teachers’ beliefs for teaching students with ASD.
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11. Mathew NE, Burton KLO, Schierbeek A, Crncec R, Walter A, Eapen V. {{Parenting preschoolers with autism: Socioeconomic influences on wellbeing and sense of competence}}. {World J Psychiatry};2019 (Mar 27);9(2):30-46.
BACKGROUND: Previous research suggests that parents raising a child with autism experience higher levels of psychological distress than parents of typically developing children and parents of children with other developmental disorders. Little is known, however, about the intersection between the effects of socioeconomic status (SES) on the wellbeing and sense of parental competency of parents of pre-schoolers with autism and how it relates to child symptom severity. AIM: To examine the relationship between their child’s symptom severity, SES, as measured by neighbourhood advantage and occupational status, on the psychological wellbeing and perceived parenting competence among parents of preschoolers with autism. METHODS: Parents of 117 preschool-aged children with a diagnosis of autism spectrum disorder (ASD), 107 mothers and 54 fathers, completed questionnaires about their child’s symptoms of ASD and functioning, their own perceptions of their wellbeing and parental competence on entry to an early intervention program in Sydney, Australia. Parents also provided demographic information pertaining to their occupation, level of education attained and address (postcode). All children were also assessed for their severity of symptoms using the Autism Diagnostic Observation Schedule. The Australian Socioeconomic Index of occupational status as a measure of familial SES and the Index of Relative Socio-economic Advantage and Disadvantage as a measure of neighbourhood advantage were used to examine the impact of SES on parental sense of competence and wellbeing. RESULTS: Compared to normative populations, both mothers and fathers in our sample reported significantly higher levels of parenting sense of efficacy but lower levels of interest in the parenting role. Mothers also displayed higher levels of satisfaction. Both mothers and fathers displayed higher levels of depression than normative populations with mothers also reporting greater levels of stress and anxiety. Child symptom severity was associated with maternal parenting competency with these relationships amplified among mothers with higher familial SES and who lived in areas of greater neighbourhood advantage. Increased adaptive functioning was associated with better maternal wellbeing, particularly among mothers who lived in areas of greater neighbourhood advantage. Contrastingly, paternal parenting competence was generally not influenced by child adaptive functioning or symptom severity, although for those in higher familial SES brackets, children’s symptom severity and maladaptive symptoms were negatively related to paternal sense of parenting efficacy. There was a trend towards moderate relationships between lower familial SES and greater depression, stress and anxiety among fathers, but no relationship with their child’s ASD symptom severity or functioning. CONCLUSION: SES differentially impacts wellbeing and sense of parenting competence and its relationship to the impact of child symptoms for mothers and fathers of preschoolers with autism.
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12. Medavarapu S, Marella LL, Sangem A, Kairam R. {{Where is the Evidence? A Narrative Literature Review of the Treatment Modalities for Autism Spectrum Disorders}}. {Cureus};2019 (Jan 16);11(1):e3901.
The most important thing about autism spectrum disorder (ASD) is that there is, in fact, no cure for this disorder; however, currently, there are many claims of pharmacological and dietary therapies and behavioral interventions that are said to improve outcome or even lead to « cure » or « recovery. » It continues to remain a challenging condition for children and their families. Research conducted on many of these treatment modalities is limited and, consequently, sufficient evidence does not exist to support their use. The primary aim of this paper was to search for the evidence of the efficacy of each treatment for autism till now. We reviewed different treatment modalities and randomized clinical trials on each treatment to look for the evidence. Although there are interventions that may be effective in alleviating some symptoms and improving skills that help autistic persons lead more productive lives, proven benefits were observed only with applied behavioral analysis (ABA) and some psychopharmacologic agents.
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13. Ortiz-Mantilla S, Cantiani C, Shafer VL, Benasich AA. {{Minimally-verbal children with autism show deficits in theta and gamma oscillations during processing of semantically-related visual information}}. {Sci Rep};2019 (Mar 25);9(1):5072.
To acquire language, children must build phonemic representations of their native language, learn to associate auditory words to visual objects and assemble a lexicon. It is not clear however, whether the limited linguistic ability seen in minimally-verbal (MV) children with Autism Spectrum Disorder (ASD) relates to deficits in cortical representation of an object and/or in linking an object to its semantic information. This EEG-based study investigated neural mechanisms underlying visual processing of common objects in MV-ASD and control children. Ten MV-ASD children, 4- to 7- years-old and 15 age/gender-matched controls, were presented with a picture-word matching paradigm. Time-frequency analyses were conducted at the sources generating the event-related responses at both early and late visual processing. Permutation testing identified spectral power and phase coherence clusters that significantly differed between the groups. As compared to controls, MV-ASD children exhibited smaller amplitudes and longer source latencies; decreased gamma and theta power with less theta phase coherence in occipital regions, and reduced frontal gamma power. Our results confirm that visual processing is altered in MV-ASD children and suggest that some of the linguistic differences observed in these children arise from impaired object/label cortical representations and reduced allocation of attention, which would impact lexical acquisition.
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14. Soda T, Mapelli L, Locatelli F, Botta L, Goldfarb M, Prestori F, D’Angelo E. {{Hyperexcitability and Hyperplasticity Disrupt Cerebellar Signal Transfer in the IB2 KO Mouse Model of Autism}}. {J Neurosci};2019 (Mar 27);39(13):2383-2397.
Autism spectrum disorders (ASDs) are pervasive neurodevelopmental conditions that often involve mutations affecting synaptic mechanisms. Recently, the involvement of cerebellum in ASDs has been suggested, but the underlying functional alterations remained obscure. We investigated single-neuron and microcircuit properties in IB2 (Islet Brain-2) KO mice of either sex. The IB2 gene (chr22q13.3 terminal region) deletion occurs in virtually all cases of Phelan-McDermid syndrome, causing autistic symptoms and a severe delay in motor skill acquisition. IB2 KO granule cells showed a larger NMDA receptor-mediated current and enhanced intrinsic excitability, raising the excitatory/inhibitory balance. Furthermore, the spatial organization of granular layer responses to mossy fibers shifted from a « Mexican hat » to a « stovepipe hat » profile, with stronger excitation in the core and weaker inhibition in the surround. Finally, the size and extension of long-term synaptic plasticity were remarkably increased. These results show for the first time that hyperexcitability and hyperplasticity disrupt signal transfer in the granular layer of IB2 KO mice, supporting cerebellar involvement in the pathogenesis of ASD.SIGNIFICANCE STATEMENT This article shows for the first time a complex set of alterations in the cerebellum granular layer of a mouse model [IB2 (Islet Brain-2) KO] of autism spectrum disorders. The IB2 KO in mice mimics the deletion of the corresponding gene in the Phelan-McDermid syndrome in humans. The changes reported here are centered on NMDA receptor hyperactivity, hyperplasticity, and hyperexcitability. These, in turn, increase the excitatory/inhibitory balance and alter the shape of center/surround structures that emerge in the granular layer in response to mossy fiber activity. These results support recent theories suggesting the involvement of cerebellum in autism spectrum disorders.
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15. Stadnick NA, Meza RD, Suhrheinrich J, Aarons GA, Brookman-Frazee L, Lyon AR, Mandell DS, Locke J. {{Leadership profiles associated with the implementation of behavioral health evidence-based practices for autism spectrum disorder in schools}}. {Autism};2019 (Mar 27):1362361319834398.
Implementation of evidence-based practice (EBP) for autism spectrum disorder (ASD) in the education system is a public health priority. Leadership is a critical driver of EBP implementation but little is known about the types of leadership behaviors exhibited by school leaders and how this influences the context of EBP implementation, particularly for students with ASD. The objectives of this study were to determine (1) the leadership profiles of principals involved in EBP implementation for students with ASD and (2) how these leadership profiles related to school characteristics and implementation climate. The Exploration, Preparation, Implementation, Sustainment (EPIS) framework was used to guide the design and analysis of this study. Participants (n = 296) included principals, teachers, and classroom support staff. They provided demographic information and completed the Multifactor Leadership Questionnaire and Implementation Climate Scale. Using latent profile analysis, a three-pattern solution was identified: Disengaged (6% of sample), Undifferentiated (23% of sample), and Optimal (71% of sample). Principals in schools with higher proportions of students with an individualized education program were more likely to be classified as Undifferentiated than Optimal. The Optimal group was associated with more positive implementation climate than the Undifferentiated or Disengaged groups. Findings suggest that leadership behaviors rated by principals and their staff involved in implementation of common autism EBPs can be meaningfully clustered into three discernible profiles that are shaped by organizational context and linked to strategic implementation climate. Our study findings have implications for leadership training and service delivery in schools by underscoring the critical nature of school leadership during implementation of EBPs for children with autism and the interplay between specific leadership behaviors and strategic implementation climate.
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16. Stanek KR, Youngkin EM, Pyle LL, Raymond JK, Driscoll KA, Majidi S. {{Prevalence, Characteristics and Diabetes Management in Children with Comorbid Autism Spectrum Disorder and Type 1 Diabetes}}. {Pediatr Diabetes};2019 (Mar 26)
OBJECTIVE: To determine autism spectrum disorder (ASD) prevalence within our pediatric type 1 diabetes (T1D) clinic population and determine clinical characteristics and technology use by individuals with both ASD and T1D compared to matched controls with T1D alone and compared to our overall pediatric T1D clinic. RESEARCH DESIGN AND METHODS: Medical chart review revealed 30 individuals with both ASD and type 1 diabetes (ASD+T1D). Controls (n=90) were matched for age, sex, race/ethnicity, and T1D duration. ASD+T1D was compared to both matched controls and the pediatric T1D clinic population. RESULTS: ASD prevalence in the pediatric T1D population was 1.16% (CI 0.96-1.26). Compared to the T1D clinic, ASD+T1D had more males (93% vs 52%; p<0.0001), lower hemoglobin A1c (HbA1c) (8.2% vs 8.9%; 66 vs 74 mmol/mol; p=0.006), and lower insulin pump (CSII) use (37% vs 56%; p<0.0001). No differences were found between ASD+T1D and matched controls in HbA1c or blood glucose checks per day. The ASD+T1D group was less likely to use CSII than matched controls (37% vs 61%; p=0.03). HbA1c did not change after CSII initiation in ASD+T1D, but increased for matched controls. CONCLUSIONS: Prevalence of ASD in the pediatric T1D population is comparable to the general population in Colorado. Individuals with ASD may experience barriers limiting CSII use, but achieve equivalent glycemic control compared to those without ASD. CSII may be more effective in maintaining lower HbA1c over time in those with ASD than in those without ASD. This article is protected by copyright. All rights reserved. Lien vers le texte intégral (Open Access ou abonnement)
17. Tamm L, Duncan A, Vaughn A, McDade R, Estell N, Birnschein A, Crosby L. {{Academic Needs in Middle School: Perspectives of Parents and Youth with Autism}}. {J Autism Dev Disord};2019 (Mar 25)
Youth with autism spectrum disorders (ASD) without intellectual disability frequently experience academic problems, in part due to executive functioning (EF) deficits. There are currently no evidence-based interventions targeting academic EF skills for middle school youth with ASD. An intervention is currently in development. This paper reports on a « proof of concept » uncontrolled trial of the intervention, and focus groups with parents and youth to inform tailoring and adaptation of the intervention. Results of the trial suggest high feasibility/satisfaction, but a need for further adaptation to promote uptake by youth with ASD. Results from the focus groups confirmed the need for an intervention targeting academic EF skills, successful strategies in use, and the need to promote increased youth independence.
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18. Tesfaye R, Courchesne V, Yusuf A, Savion-Lemieux T, Singh I, Shikako-Thomas K, Mirenda P, Waddell C, Smith IM, Nicholas D, Szatmari P, Bennett T, Duku E, Georgiades S, Kerns C, Vaillancourt T, Zaidman-Zait A, Zwaigenbaum L, Elsabbagh M. {{Assuming ability of youth with autism: Synthesis of methods capturing the first-person perspectives of children and youth with disabilities}}. {Autism};2019 (Mar 27):1362361319831487.
Most research regarding youth with autism spectrum disorder has not focused on their first-person perspectives providing limited insight into methodologies best suited to eliciting their voices. We conducted a synthesis of methods previously used to obtain the first-person perspectives of youth with various disabilities, which may be applicable to youth with autism spectrum disorder. Two-hundred and eighty-four articles met the inclusion criteria of our scoping review. We identified six distinct primary methods (questionnaires, interviews, group discussion, narratives, diaries, and art) expressed through four communication output modalities (language, sign language and gestures, writing, and images). A group of parents who have children with autism spectrum disorder were then presented with a synthesis of results. This parent consultation was used to build on approaches identified in the literature. Parents identified barriers that may be encountered during participant engagement and provided insights on how best to conduct first-person research with youth with autism spectrum disorder. Based on our findings, we present a novel methodological framework to capture the perspectives of youth with various communication and cognitive abilities, while highlighting family, youth, and expert contributions.
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19. Zaslavsky K, Zhang WB, McCready FP, Rodrigues DC, Deneault E, Loo C, Zhao M, Ross PJ, El Hajjar J, Romm A, Thompson T, Piekna A, Wei W, Wang Z, Khattak S, Mufteev M, Pasceri P, Scherer SW, Salter MW, Ellis J. {{SHANK2 mutations associated with autism spectrum disorder cause hyperconnectivity of human neurons}}. {Nat Neurosci};2019 (Apr);22(4):556-564.
Heterozygous loss-of-function mutations in SHANK2 are associated with autism spectrum disorder (ASD). We generated cortical neurons from induced pluripotent stem cells derived from neurotypic and ASD-affected donors. We developed sparse coculture for connectivity assays where SHANK2 and control neurons were differentially labeled and sparsely seeded together on a lawn of unlabeled control neurons. We observed increases in dendrite length, dendrite complexity, synapse number, and frequency of spontaneous excitatory postsynaptic currents. These findings were phenocopied in gene-edited homozygous SHANK2 knockout cells and rescued by gene correction of an ASD SHANK2 mutation. Dendrite length increases were exacerbated by IGF1, TG003, or BDNF, and suppressed by DHPG treatment. The transcriptome in isogenic SHANK2 neurons was perturbed in synapse, plasticity, and neuronal morphogenesis gene sets and ASD gene modules, and activity-dependent dendrite extension was impaired. Our findings provide evidence for hyperconnectivity and altered transcriptome in SHANK2 neurons derived from ASD subjects.
