1. Bar-Lev Schleider L, Mechoulam R, Saban N, Meiri G, Novack V. {{Real life Experience of Medical Cannabis Treatment in Autism: Analysis of Safety and Efficacy}}. {Sci Rep}. 2019; 9(1): 200.
There has been a dramatic increase in the number of children diagnosed with autism spectrum disorders (ASD) worldwide. Recently anecdotal evidence of possible therapeutic effects of cannabis products has emerged. The aim of this study is to characterize the epidemiology of ASD patients receiving medical cannabis treatment and to describe its safety and efficacy. We analysed the data prospectively collected as part of the treatment program of 188 ASD patients treated with medical cannabis between 2015 and 2017. The treatment in majority of the patients was based on cannabis oil containing 30% CBD and 1.5% THC. Symptoms inventory, patient global assessment and side effects at 6 months were primary outcomes of interest and were assessed by structured questionnaires. After six months of treatment 82.4% of patients (155) were in active treatment and 60.0% (93) have been assessed; 28 patients (30.1%) reported a significant improvement, 50 (53.7%) moderate, 6 (6.4%) slight and 8 (8.6%) had no change in their condition. Twenty-three patients (25.2%) experienced at least one side effect; the most common was restlessness (6.6%). Cannabis in ASD patients appears to be well tolerated, safe and effective option to relieve symptoms associated with ASD.
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2. Crowell JA, Keluskar J, Gorecki A. {{Parenting behavior and the development of children with autism spectrum disorder}}. {Comprehensive psychiatry}. 2018; 90: 21-9.
Autism spectrum disorders (ASD) are neurodevelopmental disorders in which multiple genetic and environmental factors play roles. Symptoms of deficits in social communication and restrictive, repetitive behavioral patterns emerge early in a child’s development. While parents do not cause these difficulties, impairments in social relatedness can strain parent child interactions and parental stress can have negative transactional effects that impede children development. Conversely, as with typically developing children, parental behavior can also enhance development in ASD and parents play a role in many interventions. In this review we examine parental contributions to the development of children with ASD, focusing on social communication and emotion regulation. We address parent and family characteristics that may impede development so they can be identified in families and interventions developed to target them.
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3. Hofer J, Bachmann C, Kamp-Becker I, Poustka L, Roessner V, Stroth S, Wolff N, Hoffmann F. {{Willingness to try and lifetime use of complementary and alternative medicine in children and adolescents with autism spectrum disorder in Germany: A survey of parents}}. {Autism}. 2019: 1362361318823545.
Regardless of their limited evidence and potential adverse effects, use of complementary and alternative medicine is common in children with autism spectrum disorder. Nevertheless, data on complementary and alternative medicine use in children with autism spectrum disorder in Germany are lacking. Therefore, a questionnaire survey on the use of complementary and alternative medicine was distributed to parents of children with autism spectrum disorder from three academic autism spectrum disorder outpatient clinics in Germany. Of 211 respondents, 46% stated that their child currently used or had ever used some form of complementary and alternative medicine in their life. The complementary and alternative medicine modalities most frequently used were manipulative and body-based methods (e.g. craniosacral therapy). And 18% of caregivers expressed willingness to try complementary and alternative medicine treatments for their child with autism spectrum disorder in the future, with mind-body interventions predominating. Health professionals should be aware of the considerable complementary and alternative medicine use prevalence among children with autism spectrum disorder and offer parents information about its effectiveness and potential side effects.
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4. Hughes RB, Robinson-Whelen S, Raymaker D, Lund EM, Oschwald M, Katz M, Starr A, Ashkenazy E, Powers LE, Nicolaidis C. {{The relation of abuse to physical and psychological health in adults with developmental disabilities}}. {Disability and health journal}. 2019.
BACKGROUND: People with developmental disabilities are at disproportionately high risk of abuse. Although considerable evidence exists on the health-related consequences of abuse in the general population, little is known about those consequences in people with developmental disabilities. OBJECTIVE: To examine the relation of abuse with psychological and physical health outcomes in adults with developmental disabilities. METHODS: We used an accessible audio computer-assisted self-interview to collect anonymous data on demographic and disability characteristics, childhood and adult abuse experiences, and physical and psychological health from 350 women and men with developmental disabilities. Abuse experience was reflected by five factor scores consisting of three child abuse factors (childhood sexual abuse, childhood physical abuse, childhood disability-related abuse) and two adult abuse factors (adult sexual abuse, adult mixed abuse). We examined each of four health outcomes (depression, post trraumatic stress disorder, physical health symptoms, secondary health conditions) separately to determine the extent to which childhood and adult abuse experiences uniquely predicted psychological and physical health outcomes above and beyond demographic and disability-related characteristics. RESULTS: All five abuse factor scores were significantly related to all four health outcomes. When examined simultaneously, childhood disability-related abuse and adult mixed abuse accounted for unique variance in outcomes. Exploratory analyses revealed no difference in the impact of abuse by gender. CONCLUSIONS: In this study, childhood disability-related abuse and adult mixed abuse significantly predicted lower levels of psychological and physical health in a sample of adults with developmental disabilities. Our findings highlight the importance of addressing abuse and its sequalae in the developmental disabilities community.
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5. Kimura T, Kagami M, Matsubara K, Yatsuga S, Mukasa R, Yatsuga C, Matsumoto T, Koga Y. {{Temple syndrome diagnosed in an adult patient with clinical autism spectrum disorder}}. {Clin Case Rep}. 2019; 7(1): 15-8.
Temple syndrome (TS14) leads to growth failure, precocious puberty, and diabetes mellitus. However, the long-term prognosis, including the development of social behavior in TS14 patients, remains unclarified. We report the clinical course of a male patient with autism spectrum disorder that received a diagnosis of TS14 at 33 years of age.
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6. Moore SM, Seidman JS, Ellegood J, Gao R, Savchenko A, Troutman TD, Abe Y, Stender J, Lee D, Wang S, Voytek B, Lerch JP, Suh H, Glass CK, Muotri AR. {{Setd5 haploinsufficiency alters neuronal network connectivity and leads to autistic-like behaviors in mice}}. {Translational psychiatry}. 2019; 9(1): 24.
SETD5, a gene linked to intellectual disability (ID) and autism spectrum disorder (ASD), is a member of the SET-domain family and encodes a putative histone methyltransferase (HMT). To date, the mechanism by which SETD5 haploinsufficiency causes ASD/ID remains an unanswered question. Setd5 is the highly conserved mouse homolog, and although the Setd5 null mouse is embryonic lethal, the heterozygote is viable. Morphological tracing and multielectrode array was used on cultured cortical neurons. MRI was conducted of adult mouse brains and immunohistochemistry of juvenile mouse brains. RNA-Seq was used to investigate gene expression in the developing cortex. Behavioral assays were conducted on adult mice. Setd5(+/-) cortical neurons displayed significantly reduced synaptic density and neuritic outgrowth in vitro, with corresponding decreases in network activity and synchrony by electrophysiology. A specific subpopulation of fetal Setd5(+/-) cortical neurons showed altered gene expression of neurodevelopment-related genes. Setd5(+/-) animals manifested several autism-like behaviors, including hyperactivity, cognitive deficit, and altered social interactions. Anatomical differences were observed in Setd5(+/-) adult brains, accompanied by a deficit of deep-layer cortical neurons in the developing brain. Our data converge on a picture of abnormal neurodevelopment driven by Setd5 haploinsufficiency, consistent with a highly penetrant risk factor.
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7. Park SH, Song YJC, Demetriou EA, Pepper KL, Norton A, Thomas EE, Hickie IB, Hermens DF, Glozier N, Guastella AJ. {{Disability, functioning, and quality of life among treatment-seeking young autistic adults and its relation to depression, anxiety, and stress}}. {Autism}. 2019: 1362361318823925.
In this study, we consecutively recruited treatment-seeking young autistic adults without intellectual impairment aged 16-30 years who presented to a mental health service and evaluated general health (distress, quality of life, and disability), functioning (work loss days and social functioning), and mood symptoms (depression, anxiety, and stress) in those diagnosed with autism spectrum disorder ( n = 96). This group was compared to young adults presenting to the same service with primary mental health disorders (depression, n = 343; bipolar, n = 132; psychosis, n = 166; and anxiety, n = 303). This study also investigated the influence of mood symptoms on general health and functioning in the autism spectrum disorder group. Young autistic adults reported significant general health and functioning impairments that were of similar degree to those presenting with primary mental health disorders. Interestingly, the autistic group also reported similarly high levels of mood symptoms to those with primary depressive and anxiety disorders. In the autistic group, depressive symptoms were strongly associated with distress, quality of life, and work loss days, while stress symptoms were strongly associated with disability. This study highlights further research, and mental health services are required specifically targeting young autistic adults to address their significant unmet needs.
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8. Schmitt LM, Shaffer RC, Hessl D, Erickson C. {{Executive Function in Fragile X Syndrome: A Systematic Review}}. {Brain Sci}. 2019; 9(1).
Executive function (EF) supports goal-directed behavior and includes key aspects such as working memory, inhibitory control, cognitive flexibility, attention, processing speed, and planning. Fragile X syndrome (FXS) is the leading inherited monogenic cause of intellectual disability and is phenotypically characterized by EF deficits beyond what is expected given general cognitive impairments. Yet, a systematic review of behavioral studies using performance-based measures is needed to provide a summary of EF deficits across domains in males and females with FXS, discuss clinical and biological correlates of these EF deficits, identify critical limitations in available research, and offer suggestions for future studies in this area. Ultimately, this review aims to advance our understanding of the underlying pathophysiological mechanisms contributing to EF in FXS and to inform the development of outcome measures of EF and identification of new treatment targets in FXS.
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9. Sutton JE, Huws JC, Burton CR. {{Sleep hygiene education and children with developmental disabilities: Findings from a co-design study}}. {Journal of intellectual disabilities : JOID}. 2019: 1744629518818950.
This qualitative study develops a programme theory demonstrating the complexity embedded in sleep hygiene education (SHE) as an intervention to improve sleep problems in children with developmental disabilities. In co-design workshops, eight parents and six sleep practitioners deliberated themes developed from findings of an earlier exploratory study of stakeholder perceptions of SHE. A SHE tool underpinned by programme theory was developed evidenced by mid-range theories of change. Analytical themes were developed to explain the programme theory and the complexities of a successful SHE intervention: the need to legitimize children’s sleep problems and consider the nature of customization, knowledge sharing, health expectation and impact of sleep service rationing and gaming strategies on implementation success. Policy and practice implications include a need to raise the public profile of children’s sleep problems and promote parental involvement in intervention implementation. Further research is needed to test out this theory-driven framework for evaluating SHE.
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10. Traynor JM, Gough A, Duku E, Shore DI, Hall GBC. {{Eye Tracking Effort Expenditure and Autonomic Arousal to Social and Circumscribed Interest Stimuli in Autism Spectrum Disorder}}. {J Autism Dev Disord}. 2019.
The social communicative deficits and repetitive behaviours seen in Autism Spectrum Disorder (ASD) may be affected by altered stimulus salience and reward attribution. The present study used eye tracking and a behavioural measure to index effort expenditure, arousal, and attention, during viewing of images depicting social scenes and subject-specific circumscribed interests in a group of 10 adults with ASD (mean age 25.4 years) and 19 typically-developing controls (mean age 20.7 years) Split-plot and one-way repeated measures ANOVAs were used to explore results. A significant difference between the ASD and control group was found in the amount of effort expended to view social and circumscribed images. The ASD group also displayed significant differences in pupillary response to social and circumscribed images, indicative of changes in autonomic arousal. Overall, the results support the social motivation hypothesis in ASD (Chevallier et al., Trends Cogn Sci 16(4):231-239, 2012) and suggest a role for autonomic arousal in the ASD symptom dyad.
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11. Uren J, Richdale AL, Cotton SM, Whitehouse AJO. {{Sleep problems and anxiety from 2 to 8 years and the influence of autistic traits: a longitudinal study}}. {Eur Child Adolesc Psychiatry}. 2019.
Whether or not childhood sleep problems and anxiety occur simultaneously, or one precedes the other, and any effect of autistic traits on this relationship remains unclear. We investigated longitudinal associations between sleep and anxiety at 2 years and sleep and anxiety at 8 years controlling for demographic variables. We also examined the additional influence of autistic traits at 2 years on sleep problems and anxiety at 8 years. Participants were from the Western Australian Pregnancy Cohort (Raine) Study, where 2900 pregnant women were recruited between 1989 and 1991 and their children assessed every 2-3 years thereafter. Demographic information was provided at 16-18 weeks gestation. Children’s sleep and anxiety at 2 and 8 years and autistic traits at 2 years were measured using the Child Behavior Checklist. Hierarchical multiple regression models tested the prediction of both anxiety and sleep problems at 8 years. Sleep problems at 2 years and 8 years, anxiety at 2 years, and autistic traits at 2 years were significantly associated with anxiety at 8 years. Sleep problems at 2 years and anxiety at 8 years were significantly related to sleep problems at 8 years. Each of these models explained about 20% of variance. Childhood sleep problems, anxiety and autistic traits are interrelated and can occur concurrently in young children, but the best predictor of poor sleep in middle childhood is concurrent anxiety and vice versa. Anxiety and sleep problems may be an early indicator of autism in young children and early autistic traits may also contribute to anxiety problems later in childhood.
