1. Agelink van Rentergem JA, Lever AG, Geurts HM. {{Negatively phrased items of the Autism Spectrum Quotient function differently for groups with and without autism}}. {Autism};2019 (Feb 28):1362361319828361.
The Autism Spectrum Quotient is a widely used instrument for the detection of autistic traits. However, the validity of comparisons of Autism Spectrum Quotient scores between groups may be threatened by differential item functioning. Differential item functioning entails a bias in items, where participants with equal values of the latent trait give different answers because of their group membership. In this article, items of the Autism Spectrum Quotient were studied for differential item functioning between different groups within a single sample ( N = 408). Three analyses were conducted. First, using a Rasch mixture model, two latent groups were detected that show differential item functioning. Second, using a Rasch regression tree model, four groups were found that show differential item functioning: men without autism, women without autism, people 50 years and younger with autism, and people older than 50 years with autism. Third, using traditional methods, differential item functioning was detected between groups with and without autism. Therefore, group comparisons with the Autism Spectrum Quotient are at risk of being affected by bias. Eight items emerged that consistently show differences in response tendencies between groups across analyses, and these items were generally negatively phrased. Two often-used short forms of the Autism Spectrum Quotient, the AQ-28 and AQ-10, may be more suitable for group comparisons.
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2. Cribb S, Kenny L, Pellicano E. {{‘I definitely feel more in control of my life’: The perspectives of young autistic people and their parents on emerging adulthood}}. {Autism};2019 (Feb 28):1362361319830029.
Long-term outcomes studies often paint a discouraging picture of the lives lived by autistic adults. Yet, their outcomes are often measured against normative markers of traditional adult roles, which may not apply to autistic people making the transition to adulthood. Here, we investigated the transition experiences of a group of young autistic people who were followed from childhood. Twenty-six young people and their parents ( n = 28) participated in semistructured interviews on the process of transition and their aspirations for the future. Parents often voiced serious concerns about the ongoing support their children would require and the severe lack of services designed to support them as adults. Yet, overall, young people reported feeling more in control of their own lives, including developing a sense of identity and personal autonomy, both of which may be rooted in young autistic people’s executive skills and their ability to develop and maintain trusting relationships with others – two potential candidate areas for targeted support. These results call into question whether the traditional standards to which we often hold young autistic people are developmentally appropriate and suggest that the pressures of striving towards more normative ways of engaging in the world may be detrimental to their well-being.
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3. Duvall SW, Huang-Storms L, Presmanes Hill A, Myers J, Fombonne E. {{No Sex Differences in Cognitive Ability in Young Children with Autism Spectrum Disorder}}. {J Autism Dev Disord};2019 (Feb 27)
Inconsistent findings regarding sex differences in cognition have been found in people with autism spectrum disorder (ASD). This study evaluated sex differences in cognitive-developmental functioning in a large clinical sample of young children diagnosed with ASD. The sample included children 18-68 months of age who received the Mullen Scales of Early Learning (MSEL) through Autism Treatment Network (ATN) sites from 2007 to 2013 (N = 1587, 16.7% female). In this large clinically referred sample of young children with ASD in the United States, no significant differences were found between the sexes for the MSEL Early Learning Composite (ELC) standard score, domain T Scores or age equivalents. These findings persisted when examining different age ranges, cognitive levels and domain profiles.
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4. Germone MM, Gabriels RL, Guerin NA, Pan Z, Banks T, O’Haire ME. {{Animal-assisted activity improves social behaviors in psychiatrically hospitalized youth with autism}}. {Autism};2019 (Feb 28):1362361319827411.
There is preliminary research suggesting that animal-assisted activities can improve social interactions of children with autism spectrum disorder. This pilot study sought to investigate the benefits of animal-assisted activities with dogs and psychiatrically hospitalized youth with autism spectrum disorder. Participants were recruited from a specialized inpatient psychiatric hospital unit for youth with autism spectrum disorder and other developmental disabilities. Utilizing a crossover design, participants served as their own control by engaging in two 10-min conditions: an experimental dog and handler interaction (animal-assisted activities) and a novel toy and handler control (control). Of the 142 youth aged 6–8 years screened for participation, 47 completed both conditions. Participants’ behavioral data were captured via video and coded using the Observation of Human-Animal Interaction for Research, a tool specifically developed to capture human behavioral interactions in the presence of animals. Overall, social-communication behaviors significantly improved in the animal-assisted activities experimental condition compared to the control condition ( p = 0.0001). Specifically, participants in the animal-assisted activities experimental condition displayed more positive emotional facial expressions ( p 0.0001), talking ( p = 0.0408), use of gestures ( p = 0.032), and looking at both adults and peers ( p 0.0001). In addition, a higher frequency of constant motion ( p = 0.003) was observed in the animal-assisted activities experimental condition. Results suggest that animal-assisted activities with a dog may promote social-communication behaviors in psychiatrically hospitalized youth with autism spectrum disorder. Given the fact that social and communication behaviors can facilitate treatment engagement for this population, we recommend future studies examine how such improvements can positively affect the psychiatric treatment of this population.
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5. Htut M, Ho E, Wiles J. {{A Study of Asian Children Who are Diagnosed with Autism Spectrum Disorder and Available Support Services in Auckland, New Zealand}}. {J Autism Dev Disord};2019 (Feb 28)
This study reviews the demographic characteristics of Asian children diagnosed with autism spectrum disorder (ASD) in Auckland, New Zealand, the support services they can access, and how more equitable access to health services can be provided. We examined government and non-government support services for Asian children diagnosed with ASD and their families. The findings reflect the complexities of navigating and accessing health, disability, education and social support services. Analysis of Ministry of Health Disability Support Services (DSSs) data suggest that Asians in New Zealand are underrepresented in utilizing DSSs. Drawing on Andersen’s health care utilization model, suggestions for more equitable access to health, disability, education and social support include culturally and linguistically appropriate health care and social policies.
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6. Huang F, Chen X, Jiang X, Niu J, Cui C, Chen Z, Sun J. {{Betaine Ameliorates Prenatal Valproic Acid-induced Autism-like Behavioral Abnormalities in Mice by Promoting Homocysteine Metabolism}}. {Psychiatry Clin Neurosci};2019 (Feb 28)
AIM: Abnormal high level of Homocysteine (Hcy) is associated with autism spectrum disorder (ASD). Betaine is a methyl group donor in Hcy metabolism, and is known to prevent noxious Hcy accumulation. This study explored whether betaine could influence Hcy metabolism in a mouse model of autism and ameliorate behavioral abnormalities. METHODS: Pregnant ICR mice were administered intraperitoneally with valproic acid (VPA) at E12.5. Serum Hcy concentrations in the offspring were measured by ELISA. Expressions of Hcy metabolism related enzymes, BHMT, CBS and MTR, were measured by qRT-PCR and western blotting. Offspring were treated by either betaine or saline at the age of 8 weeks and serum Hcy concentrations were measured. Social behaviors were assessed by sniff duration test and three chamber test. Repetitive behavior was evaluated by marble burying test. Tail flick test was performed to measure nociceptive sensitivity. RESULTS: Prenatal VPA-exposed mice showed significantly elevated Hcy concentrations and decreased BHMT expression. Treatment with betaine could reduce Hcy level in VPA-exposed mice, attenuate social impairment and repetitive behavior, and normalize nociceptive sensitivity in this model. CONCLUSION: Betaine could ameliorate autism-like features and play a beneficial role in mouse autism model induced by prenatal VPA exposure. This article is protected by copyright. All rights reserved.
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7. Jalnapurkar I, Cochran DM, Frazier JA. {{New Therapeutic Options for Fragile X Syndrome}}. {Curr Treat Options Neurol};2019 (Feb 27);21(3):12.
PURPOSE OF REVIEW: The purpose of this review is to provide an overview of current research and clinical practice guidelines in fragile X syndrome (FXS) with regard to therapeutic approaches in the management of this condition. The authors summarize and discuss findings from relevant preclinical studies and results from clinical trials in human subjects with FXS. Additionally, we provide an outline of the basic framework for understanding and providing educational and psychosocial supports for these individuals. RECENT FINDINGS: Current treatments in FXS are largely symptom based and focused on managing associated psychiatric and behavioral co-morbidities. While data from animal studies has been promising in providing targeted treatments to correct the underlying deficits at the cellular level, there have not been as robust findings in human trials. There are several targeted treatments for FXS currently under development. Individuals with FXS present with several behavioral challenges including anxiety, social withdrawal, ADHD, hyperarousal, self-injury, and aggression. Therapeutic services are often necessary, such as behavioral intervention, speech and language therapy, occupational therapy, and individualized educational support; adjunctive psychopharmacologic treatment is often helpful as well. It is important to address these symptoms and weigh the evidence for the use of medications that target the underlying neurobiology and pathophysiology of the syndrome.
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8. Kapp SK, Steward R, Crane L, Elliott D, Elphick C, Pellicano E, Russell G. {{‘People should be allowed to do what they like’: Autistic adults’ views and experiences of stimming}}. {Autism};2019 (Feb 28):1362361319829628.
‘Stereotyped or repetitive motor movements’ are characterised as core features in the diagnosis of autism, yet many autistic adults (and the neurodiversity movement) have reclaimed them as ‘stimming’. Supported by a growing body of scientific research, autistic adults argue that these behaviours may serve as useful coping mechanisms, yet little research has examined stimming from the perspective of autistic adults. Through interviews and focus groups, we asked 32 autistic adults to share their perceptions and experiences of stimming, including the reasons they stim, any value doing so may hold for them and their perceptions of others’ reactions to stimming. Using thematic analysis, we identified two themes: stimming as (1) a self-regulatory mechanism and (2) lacking in social acceptance, but can become accepted through understanding. Autistic adults highlighted the importance of stimming as an adaptive mechanism that helps them to soothe or communicate intense emotions or thoughts and thus objected to treatment that aims to eliminate the behaviour.
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9. Paysour MJ, Bolte AC, Lukens JR. {{Crosstalk Between the Microbiome and Gestational Immunity in Autism-Related Disorders}}. {DNA Cell Biol};2019 (Feb 28)
The etiologies of most neurodevelopmental disorders, including autism spectrum disorder (ASD), remain incompletely understood. However, recent epidemiological and experimental data suggest that dysregulated maternal immune activation (MIA) can impede normal brain maturation and promote the development of autism-related phenotypes. Indeed, our studies and work by others demonstrate that offspring born to pregnant animals that were exposed to immune activators develop many of the defining behavioral features of ASD, including abnormalities in social preference, communicative impairments, and repetitive/stereotyped behaviors. Although mounting evidence implicates key roles for hyperactive gestational inflammatory responses in neurodevelopmental disorders, the specific immune pathways that provoke autism-related phenotypes remain poorly described. The microbiome is recognized as a key modulator of immune responses, and emerging studies suggest that microbiota composition is a pivotal regulator of central nervous system function and disease. There has been growing speculation that changes in human microflora diversity contribute at some level to the recent rise in autism incidence. This has largely stemmed from reports of dysbiosis and gastrointestinal inflammation in autistic individuals. Given these clinical findings and the well-described role of the microbiome in calibrating the immune system, our group and others have recently become interested in investigating how changes in microbiota landscape influence neurodevelopmental disorder pathogenesis. In this review, we highlight emerging data describing roles for microbiota in the development of autism-related behavioral abnormalities. These recent findings identify the immune system as a link between gut microbiota and the brain in neurodevelopmental disorders, and suggest that targeting the microbiome and maternal immune responses during gestation may offer strategies to limit autism development in at-risk pregnancies.
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10. Perihan C, Burke M, Bowman-Perrott L, Bicer A, Gallup J, Thompson J, Sallese M. {{Effects of Cognitive Behavioral Therapy for Reducing Anxiety in Children with High Functioning ASD: A Systematic Review and Meta-Analysis}}. {J Autism Dev Disord};2019 (Feb 27)
Children with autism spectrum disorder (ASD) are at greater risk for experiencing high levels of anxiety symptoms. Recent evidence suggests Cognitive behavioral therapy (CBT) may also be effective for anxiety reduction in some presentations of ASD. This meta-analysis evaluated twenty-three studies. Results yielded a moderate effect size (g = – 0.66) for the reduction of anxiety symptoms. Moderators indicated larger effects for studies were achieved with parental involvement (g = – 0.85, p < .05) than with child-only treatments (g = - 0.34, p < .05). Short-term interventions generated a smaller effect (g = - 0.37 p < .05) than either standard-term (g = - 1.02, p < .05) or long-term interventions (g = - 0.69, p < .05).Implications for children with ASD are discussed. Lien vers le texte intégral (Open Access ou abonnement)
11. Ponson L, Gomot M, Blanc R, Barthelemy C, Roux S, Munnich A, Romana S, Aguillon-Hernandez N, Malan V, Bonnet-Brilhault F. {{Author Correction: 22q13 deletion syndrome: communication disorder or autism? Evidence from a specific clinical and neurophysiological phenotype}}. {Transl Psychiatry};2019 (Feb 28);9(1):101.
Since the online publication of the above article, the authors have noted errors with the author list. The author names were listed as ‘(last name)(first name)’ instead of ‘(first name)(last name)’.
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12. Sam AM, Cox AW, Savage MN, Waters V, Odom SL. {{Disseminating Information on Evidence-Based Practices for Children and Youth with Autism Spectrum Disorder: AFIRM}}. {J Autism Dev Disord};2019 (Feb 28)
Comprehensive reviews of the research literature have identified that focused intervention practices for children and youth with autism spectrum disorder have evidence of producing positive developmental and learning outcomes. The Autism Focused Intervention Resources and Modules (AFIRM) project has translated evidence-based practices identified by Wong et al. (Journal of Autism and Developmental Disorders 45(7):1951-1966, 2015) into online learning modules. The purpose of this paper is to describe (1) the process for translating the research literature into practical information that practitioners can use, (2) its dissemination through a freely accessible website, (3) the use of the modules by over 64,500 users located in the United States and abroad, (4) knowledge gained as a result of completing the modules, and (5) consumers’ evaluations of modules usefulness and relevance.
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13. Schmidt RJ, Iosif AM, Guerrero Angel E, Ozonoff S. {{Association of Maternal Prenatal Vitamin Use With Risk for Autism Spectrum Disorder Recurrence in Young Siblings}}. {JAMA Psychiatry};2019 (Feb 27)
Importance: Maternal use of folic acid supplements has been inconsistently associated with reduced risk for autism spectrum disorder (ASD) in the child. No study to date has examined this association in the context of ASD recurrence in high-risk families. Objective: To examine the association between maternal prenatal vitamin use and ASD recurrence risk in younger siblings of children with ASD. Design, Setting, and Participants: This prospective cohort study analyzed data from a sample of children (n = 332) and their mothers (n = 305) enrolled in the MARBLES (Markers of Autism Risk in Babies: Learning Early Signs) study. Participants in the MARBLES study were recruited at the MIND Institute of the University of California, Davis and were primarily from families receiving services for children with ASD in the California Department of Developmental Services. In this sample, the younger siblings at high risk for ASD were born between December 1, 2006, and June 30, 2015, and completed a final clinical assessment within 6 months of their third birthday. Prenatal vitamin use during pregnancy was reported by mothers during telephone interviews. Data analysis for this study was conducted from January 1, 2017, to December 3, 2018. Main Outcomes and Measures: Autism spectrum disorder, other nontypical development (non-TD), and typical development (TD) were algorithmically defined according to Autism Diagnostic Observation Schedule and Mullen Scales of Early Learning subscale scores. Results: After exclusions, the final sample comprised 241 younger siblings, of which 140 (58.1%) were male and 101 (41.9%) were female, with a mean (SD) age of 36.5 (1.6) months. Most mothers (231 [95.9%]) reported taking prenatal vitamins during pregnancy, but only 87 mothers (36.1%) met the recommendations to take prenatal vitamins in the 6 months before pregnancy. The prevalence of ASD was 14.1% (18) in children whose mothers took prenatal vitamins in the first month of pregnancy compared with 32.7% (37) in children whose mothers did not take prenatal vitamins during that time. Children whose mothers reported taking prenatal vitamins during the first month of pregnancy were less likely to receive an ASD diagnosis (adjusted relative risk [RR], 0.50; 95% CI, 0.30-0.81) but not a non-TD 36-month outcome (adjusted RR, 1.14; 95% CI, 0.75-1.75) compared with children whose mothers reported not taking prenatal vitamins. Children in the former maternal prenatal vitamin group also had statistically significantly lower autism symptom severity (adjusted estimated difference, -0.60; 95% CI, -0.97 to -0.23) and higher cognitive scores (adjusted estimated difference, 7.1; 95% CI, 1.2-13.1). Conclusions and Relevance: Maternal prenatal vitamin intake during the first month of pregnancy may reduce ASD recurrence in siblings of children with ASD in high-risk families. Additional research is needed to confirm these results; to investigate dose thresholds, contributing nutrients, and biologic mechanisms of prenatal vitamins; and to inform public health recommendations for ASD prevention in affected families.
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14. Smith DaWalt L, Hong J, Greenberg JS, Mailick MR. {{Mortality in individuals with autism spectrum disorder: Predictors over a 20-year period}}. {Autism};2019 (Feb 28):1362361319827412.
Research has shown that individuals with autism spectrum disorder have higher rates of health problems throughout childhood, adolescence, and adulthood, and that this may result in elevated risk of early mortality. This study reported the rate, timing, and causes of death in a large community-based cohort of adolescents and adults with autism spectrum disorder ( n = 406) over a 20-year period (1998-2018) and identified predictors of mortality. Over this period, 6.4% of individuals died at an average age of 39 years. Causes of death included chronic conditions (such as cancer and heart disease), accidents (such as choking on food and accidental poisoning), and health complications due to medication side effects. Even after controlling for age and health status, significant predictors of mortality were early childhood levels of impairments in social reciprocity and high levels of functional impairments at the start of the study period. The results suggest the importance of social engagement and functional self-sufficiency across the life course, as well as adequate access to health care for individuals with autism spectrum disorder.
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15. Sweetman DU, O’Donnell SM, Lalor A, Grant T, Greaney H. {{Zinc and Vitamin A Deficiency in a Cohort of Children with Autism Spectrum Disorder}}. {Child Care Health Dev};2019 (Feb 28)
BACKGROUND AND OBJECTIVES: Studies suggest that trace element and vitamin deficiencies are common in children with Autism Spectrum Disorder (ASD). Data describing the rates of vitamin and trace element deficiencies in the ASD population of the North-West of Ireland is lacking. We wished to determine the prevalence of zinc and vitamin A deficiency in the ASD population compared to controls within this geographical area. METHODS: Parents of children aged 2-18 years with ASD were invited to participate in the study. The control group consisted of well children attending the paediatric department for routine blood sampling. Children on vitamin supplements were excluded from both ASD and control groups. Informed written consent was obtained prior to recruitment. Samples were analysed for zinc and vitamin A levels according to standardised laboratory procedures. RESULTS: Seventy-four of the 150 children with ASD who were invited and 72 controls underwent blood sampling. Mean zinc and vitamin A levels were normal in both groups. There were significantly more males in the ASD group (88% versus 56%, p-value<0.001). The mean (SD) zinc level was not different between the groups [ASD 11.7 (1.7) versus control 11.6 (2.1) mumol/L, p-value = 0.86]. The mean (SD) vitamin A level was higher in the ASD group [ASD 350.6 (82.6) versus 319.2 (82.8) mug/L, p-value = 0.03] but this was likely confounded by age. CONCLUSION: Children with ASD in the North-West of Ireland have mean zinc and vitamin A levels within the normal range. It is important that these findings are relayed to health professionals and to parents of children with ASD so that informed decisions on vitamin supplementation can be made. Lien vers le texte intégral (Open Access ou abonnement)
16. Tarver J, Palmer M, Webb S, Scott S, Slonims V, Simonoff E, Charman T. {{Child and parent outcomes following parent interventions for child emotional and behavioral problems in autism spectrum disorders: A systematic review and meta-analysis}}. {Autism};2019 (Feb 28):1362361319830042.
There is growing interest in the development of behavioral parent interventions targeting emotional and behavioral problems in children with autism spectrum disorders. Such interventions have potential to improve a number of child and parental well-being outcomes beyond disruptive child behavior. This systematic review and meta-analysis assesses evidence for the efficacy of behavioral parent interventions for disruptive and hyperactive child behavior in autism spectrum disorders, as well as parenting efficacy and stress. A total of 11 articles from nine randomized controlled trials were included. Sufficient data were available to calculate standardized mean difference and show favorable effects of behavioral parent interventions on parent-reported measures of child disruptive behavior (standardized mean difference = 0.67), hyperactivity (standardized mean difference = 0.31) and parent stress (standardized mean difference = 0.37); effects on parent efficacy are less clear (standardized mean difference = 0.39, p = 0.17). There were insufficient data to explore intervention effects on internalizing behavior in autism spectrum disorders, parenting behaviors, or observational and teacher-reported outcomes, providing important avenues for future research. This review adds to growing evidence of the efficacy of behavioral parent interventions for child behavior and parental well-being in autism spectrum disorders (Prospero: CRD42016033979).
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17. Thongkorn S, Kanlayaprasit S, Jindatip D, Tencomnao T, Hu VW, Sarachana T. {{Sex Differences in the Effects of Prenatal Bisphenol A Exposure on Genes Associated with Autism Spectrum Disorder in the Hippocampus}}. {Sci Rep};2019 (Feb 28);9(1):3038.
Autism spectrum disorder (ASD) is a neurodevelopmental disorder inexplicably biased towards males. Although prenatal exposure to bisphenol A (BPA) has recently been associated with the ASD risk, whether BPA dysregulates ASD-related genes in the developing brain remains unclear. In this study, transcriptome profiling by RNA-seq analysis of hippocampi isolated from neonatal pups prenatally exposed to BPA was conducted and revealed a list of differentially expressed genes (DEGs) associated with ASD. Among the DEGs, several ASD candidate genes, including Auts2 and Foxp2, were dysregulated and showed sex differences in response to BPA exposure. The interactome and pathway analyses of DEGs using Ingenuity Pathway Analysis software revealed significant associations between the DEGs in males and neurological functions/disorders associated with ASD. Moreover, the reanalysis of transcriptome profiling data from previously published BPA studies consistently showed that BPA-responsive genes were significantly associated with ASD-related genes. The findings from this study indicate that prenatal BPA exposure alters the expression of ASD-linked genes in the hippocampus and suggest that maternal BPA exposure may increase ASD susceptibility by dysregulating genes associated with neurological functions known to be negatively impacted in ASD, which deserves further investigations.
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18. Vashi N, Justice MJ. {{Treating Rett syndrome: from mouse models to human therapies}}. {Mamm Genome};2019 (Feb 28)
Rare diseases are very difficult to study mechanistically and to develop therapies for because of the scarcity of patients. Here, the rare neuro-metabolic disorder Rett syndrome (RTT) is discussed as a prototype for precision medicine, demonstrating how mouse models have led to an understanding of the development of symptoms. RTT is caused by mutations in the X-linked gene methyl-CpG-binding protein 2 (MECP2). Mecp2-mutant mice are being used in preclinical studies that target the MECP2 gene directly, or its downstream pathways. Importantly, this work may improve the health of RTT patients. Clinical presentation may vary widely among individuals based on their mutation, but also because of the degree of X chromosome inactivation and the presence of modifier genes. Because it is a complex disorder involving many organ systems, it is likely that recovery of RTT patients will involve a combination of treatments. Precision medicine is warranted to provide the best efficacy to individually treat RTT patients.
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19. Warnell KR, Maniscalco S, Baker S, Yi R, Redcay E. {{Social and delay discounting in autism spectrum disorder}}. {Autism Res};2019 (Feb 28)
Current literature is divided over whether and how processes such as perspective taking and reward sensitivity differ between individuals with autism spectrum disorder (ASD) versus neurotypical individuals. Discounting tasks may provide novel insight into how these processes operate. In delay discounting tasks, participants choose between smaller immediate rewards and larger delayed rewards, and in social discounting tasks, participants choose between a smaller monetary rewards for themselves versus a larger reward for partners of varied social distance (e.g., a close friend vs. an acquaintance). Delay and social discounting tasks thus implicitly measure the subjective value of rewards given to one’s future self and to others, capturing constructs such as perspective taking, reward processing, and social closeness, all of which have been discussed as core cognitive mechanisms underlying ASD. Despite extensive research on discounting in other clinical populations, few studies have examined delay discounting in ASD and no research has examined social discounting in ASD. The goal of the current study was to assess delay and social discounting for monetary rewards in a single sample of adolescents and adults with ASD compared to a matched neurotypical sample. Overall, adults and adolescents with ASD valued both future rewards and rewards given to others less than their typical counterparts did, but rates of discounting were not significantly correlated across temporal and social domains. These results extend an important behavioral paradigm for understanding both perspective taking and reward processing to autism. Autism Res 2019, 00: 1-8. (c) 2019 International Society for Autism Research, Wiley Periodicals, Inc. LAY SUMMARY: Discounting tasks-which experimentally measure the subjective value of different rewards-have been used with a variety of clinical populations, but are underexplored in ASD. We found that compared to neurotypical individuals, individuals with ASD showed diminished subjective value for future rewards (compared to immediate rewards) and rewards for others (compared to rewards for self). This finding has implications for understanding perspective taking, reward processing, and social closeness in ASD.
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20. Wenzel HJ, Murray KD, Haify SN, Hunsaker MR, Schwartzer JJ, Kim K, La Spada AR, Sopher BL, Hagerman PJ, Raske C, Severijnen L, Willemsen R, Hukema RK, Berman RF. {{Astroglial-targeted expression of the fragile X CGG repeat premutation in mice yields RAN translation, motor deficits and possible evidence for cell-to-cell propagation of FXTAS pathology}}. {Acta Neuropathol Commun};2019 (Feb 26);7(1):27.
The fragile X premutation is a CGG trinucleotide repeat expansion between 55 and 200 repeats in the 5′-untranslated region of the fragile X mental retardation 1 (FMR1) gene. Human carriers of the premutation allele are at risk of developing the late-onset neurodegenerative disorder, fragile X-associated tremor/ataxia syndrome (FXTAS). Characteristic neuropathology associated with FXTAS includes intranuclear inclusions in neurons and astroglia. Previous studies recapitulated these histopathological features in neurons in a knock-in mouse model, but without significant astroglial pathology. To determine the role of astroglia in FXTAS, we generated a transgenic mouse line (Gfa2-CGG99-eGFP) that selectively expresses a 99-CGG repeat expansion linked to an enhanced green fluorescent protein (eGFP) reporter in astroglia throughout the brain, including cerebellar Bergmann glia. Behaviorally these mice displayed impaired motor performance on the ladder-rung test, but paradoxically better performance on the rotarod. Immunocytochemical analysis revealed that CGG99-eGFP co-localized with GFAP and S-100ss, but not with NeuN, Iba1, or MBP, indicating that CGG99-eGFP expression is specific to astroglia. Ubiquitin-positive intranuclear inclusions were found in eGFP-expressing glia throughout the brain. In addition, intracytoplasmic ubiquitin-positive inclusions were found outside the nucleus in distal astrocyte processes. Intriguingly, intranuclear inclusions, in the absence of eGFP mRNA and eGFP fluorescence, were present in neurons of the hypothalamus and neocortex. Furthermore, intranuclear inclusions in both neurons and astrocytes displayed immunofluorescent labeling for the polyglycine peptide FMRpolyG, implicating FMRpolyG in the pathology found in Gfa2-CGG99 mice. Considered together, these results show that Gfa2-CGG99 expression in mice is sufficient to induce key features of FXTAS pathology, including formation of intranuclear inclusions, translation of FMRpolyG, and deficits in motor function.
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21. Wyman J, Claro A. {{The UCLA PEERS School-Based Program: Treatment Outcomes for Improving Social Functioning in Adolescents and Young Adults with Autism Spectrum Disorder and Those with Cognitive Deficits}}. {J Autism Dev Disord};2019 (Feb 28)
This study examined the efficacy of the school-based Program for the Education and Enrichment of Relational Skills on the social functioning of young adults with autism and cognitive deficits. The program was administered bi-weekly at a private school for students with special needs. Students’ social etiquette knowledge, friendship engagement and teacher reported levels of their social functioning were assessed pre- and post-intervention. All participants experienced significantly improved knowledge of appropriate social etiquette. Further, students with cognitive deficits, but not those with autism, reported a significant increase in friendship engagement. Overall, the PEERS school curriculum benefited young adults with cognitive deficits, but the students with ASD experienced more challenges applying their new social skills outside of the program.
