1. Banney RM, Harper-Hill K, Arnott WL. {{The Autism Diagnostic Observation Schedule and narrative assessment: Evidence for specific narrative impairments in autism spectrum disorders}}. {International journal of speech-language pathology}. 2014 Dec 26:1-13.
Purpose: The Autism Diagnostic Observation Schedule (ADOS) contains a narrative generation task in which clients tell a story from a wordless picture book; however, the resulting narrative is not usually examined for its linguistic properties. This study aimed to examine narrative generation in autism spectrum disorder (ASD) by comparing narratives elicited from children with ASD during the ADOS to those produced by language-matched typically-developing (TD) peers. Method: Participants were children with ASD (n = 11) and TD controls (n = 17). Both groups were aged 9-15 years and were matched for expressive and receptive language skills and non-verbal intelligence. Narratives were analysed for local structure elements (length, fluency, errors, semantics and syntax), cohesion and global elements (story grammar and internal state language). Results: Results indicated that the narratives of the children with ASD were syntactically less complex, contained more ambiguous pronouns and included fewer story grammar elements than their control counterparts; with further analysis showing differences between younger and older children. Conclusions: The present findings provide evidence that children with ASD exhibit subtle story generation impairments and provide preliminary support for the inclusion of narratives elicited as part of the ADOS in the assessment of specific language skills in this population.
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2. Kryzak LA, Cengher M, Feeley KM, Fienup DM, Jones EA. {{A community support program for children with autism and their typically developing siblings: Initial investigation}}. {Journal of intellectual disabilities : JOID}. 2014 Dec 26.
Siblings are a critical part of lifelong support for individuals with autism spectrum disorder (ASD). But siblings face their own social-emotional adjustment needs. These needs may be addressed through programs that include support groups specifically for the siblings. This study examined the effects of a community program on typical siblings’ depression, anxiety, ASD knowledge, and peer network as well as reciprocal interactions between the typical sibling and sibling with ASD. The program provided a sibling support group, a skills intervention for children with ASD, and an inclusive recreation time. Siblings reported significant decreases in depression and physiological anxiety and improvements in their peer network. Autism knowledge increased but only approached significance. Direct observations revealed improvement in reciprocal interactions by most children that did not reach statistical significance. Parents, typical siblings, and interventionists indicated positive reactions to the program and its goals and outcomes. Findings are discussed in terms of the need to continue to explore interventions for siblings of children with ASD.
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3. Mitra M, Parish SL, Clements KM, Cui X, Diop H. {{Pregnancy Outcomes Among Women with Intellectual and Developmental Disabilities}}. {American journal of preventive medicine}. 2014 Dec 26.
BACKGROUND: There is currently no population-based research on the maternal characteristics or birth outcomes of U.S. women with intellectual and developmental disabilities (IDDs). Findings from small-sample studies among non-U.S. women indicate that women with IDDs and their infants are at higher risk of adverse health outcomes. PURPOSE: To describe the maternal characteristics and outcomes among deliveries to women with IDDs and compare them to women with diabetes and the general obstetric population. METHODS: Data from the 1998-2010 Massachusetts Pregnancy to Early Life Longitudinal database were analyzed between November 2013 and May 2014 to identify in-state deliveries to Massachusetts women with IDDs. RESULTS: Of the 916,032 deliveries in Massachusetts between 1998 and 2009, 703 (<0.1%) were to women with IDDs. Deliveries to women with IDDs were to those who were younger, less educated, more likely to be black and Hispanic, and less likely to be married. They were less likely to identify the father on the infant’s birth certificate, more likely to smoke during pregnancy, and less likely to receive prenatal care during the first trimester compared to deliveries to women in the control groups (p<0.01). Deliveries to women with IDDs were associated with an increased risk of adverse outcomes, including preterm delivery, very low and low birth weight babies, and low Apgar scores. CONCLUSIONS: Women with IDDs are at a heightened risk for adverse pregnancy outcomes. These findings highlight the need for a systematic investigation of the pregnancy-related risks, complications, costs, and outcomes of women with IDDs.
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4. Wegener E, Brendel C, Fischer A, Hulsmann S, Gartner J, Huppke P. {{Characterization of the MeCP2R168X Knockin Mouse Model for Rett Syndrome}}. {PloS one}. 2014;9(12):e115444.
Rett syndrome, one of the most common causes of mental retardation in females, is caused by mutations in the X chromosomal gene MECP2. Mice deficient for MeCP2 recapitulate some of the symptoms seen in patients with Rett syndrome. It has been shown that reactivation of silent MECP2 alleles can reverse some of the symptoms in these mice. We have generated a knockin mouse model for translational research that carries the most common nonsense mutation in Rett syndrome, R168X. In this article we describe the phenotype of this mouse model. In male MeCP2R168X mice life span was reduced to 12-14 weeks and bodyweight was significantly lower than in wild type littermates. First symptoms including tremor, hind limb clasping and inactivity occurred at age 27 days. At age 6 weeks nest building, rotarod, open-field and elevated plus maze experiments showed impaired motor performance, reduced activity and decreased anxiety-like behavior. Plethysmography at the same time showed apneas and irregular breathing with reduced frequency. Female MeCP2R168X mice showed no significant abnormalities except decreased performance on the rotarod at age 9 months. In conclusion we show that the male MeCP2R168X mice have a phenotype similar to that seen in MECP2 knockout mouse models and are therefore well suited for translational research. The female mice, however, have a much milder and less constant phenotype making such research with this mouse model more challenging.
