Publications by authors named "Kate Horsler"

5 Publications

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Brief Report: A Longitudinal Study of Excessive Smiling and Laughing in Children with Angelman Syndrome.

J Autism Dev Disord 2015 Aug;45(8):2624-7

Cerebra Centre for Neurodevelopmental Disorders, School of Psychology, University of Birmingham, Birmingham, B15 2TT, UK,

Elevated laughing and smiling is a key characteristic of the Angelman syndrome behavioral phenotype, with cross-sectional studies reporting changes with environment and age. This study compares levels of laughing and smiling in 12 participants across three experimental conditions [full social interaction (with eye contact), social interaction with no eye contact, proximity only] at two data points. No differences were noted in frequency of laughing and smiling over time in any condition. However, with age as a covariate, the frequency of laughing and smiling decreased over time in the full social interaction (with eye contact) condition only. As this is the first longitudinal study to explore these behaviors in Angelman syndrome, the results suggest a gene-environment-time interaction within the behavioral phenotype.
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http://dx.doi.org/10.1007/s10803-015-2404-yDOI Listing
August 2015

Age related change in social behavior in children with Angelman syndrome.

Am J Med Genet A 2011 Jun 12;155A(6):1290-7. Epub 2011 May 12.

Cerebra Centre for Neurodevelopmental Disorders, School of Psychology, University of Birmingham, UK.

We investigated the relationship between age and laughing and smiling in children with Angelman syndrome. Twenty-four children with Angelman syndrome were exposed to three experimentally manipulated conditions: proximity only, restricted social interaction, and social interaction. Children smiled the most in the social interaction condition and the least in the proximity only condition confirming the effect of social interaction on these behaviors. There was a decline in smiling and laughing in the oldest group (13.4-15.9 years) only in the social interaction condition. This trajectory of a decline in resource soliciting behaviors with age is consistent with predictions based on kinship theory.
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http://dx.doi.org/10.1002/ajmg.a.33964DOI Listing
June 2011

Experimental functional analysis of aggression in children with Angelman syndrome.

Res Dev Disabil 2009 Sep-Oct;30(5):1095-106. Epub 2009 Apr 9.

Cerebra Centre for Neurodevelopmental Disorders, School of Psychology, University of Birmingham, Birmingham, UK.

Background: Kinship theory suggests that genomic imprinting could account for phenotypic behaviors that increase (in the case of Angelman syndrome) or decrease (for Prader-Willi syndrome) the drive to access social resources (adult contact) depending on the imprinting parent-of-origin. Difficult to manage behaviors, such as aggression that is common in Angelman syndrome, could serve the function of increasing social interaction. We hypothesise that the commonly reported aggressive behavior in children with Angelman syndrome will be attention maintained.

Methods: Experimental functional analysis was carried out with twelve children with Angelman syndrome caused by either a deletion (n=10) or uniparental disomy (n=2). The relative increase and decrease of aggressive behaviors was observed in response to experimentally manipulated levels of adult attention and demand. Laughing and smiling, crying and frowning, and physical initiation with an adult were also measured.

Results: Aggression was seen in ten of the twelve children. One child evidenced a pattern of aggression across conditions consistent with maintenance by attention, three children showed higher levels of aggression during social interaction and two children showed escape motivated aggression.

Discussion: With the exception of one child the results did not confirm the hypothesis. However, the pattern of increased aggression in the high social contact condition combined with evidence of positive affect during this condition suggests aggression may serve to both maintain and initiate social contact and this interpretation is consistent with previous research. The negative results may also have been influenced by the age of the children and the low levels of observed aggression.
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http://dx.doi.org/10.1016/j.ridd.2009.03.005DOI Listing
August 2009

Genomic imprinting and the expression of affect in Angelman syndrome: what's in the smile?

J Child Psychol Psychiatry 2007 Jun;48(6):571-9

School of Psychology, University of Birmingham, UK.

Background: Kinship theory (or the genomic conflict hypothesis) proposes that the phenotypic effects of genomic imprinting arise from conflict between paternally and maternally inherited alleles. A prediction arising for social behaviour from this theory is that imbalance in this conflict resulting from a deletion of a maternally imprinted gene, as in Angelman syndrome (AS), will result in a behavioural phenotype that should evidence behaviours that increase access to maternally provided social resources (adult contact).

Method: Observation of the social behaviour of children with AS (n = 13), caused by a deletion at 15q11-q13, and a matched comparison group (n = 10) was undertaken for four hours in a socially competitive setting and the effect of adult attention on child behaviours and the effect of child smiling on adult behaviours evaluated using group comparisons and observational lag sequential analyses.

Results: The AS group smiled more than the comparison group in all settings, which had different levels of adult attention, and more when the level of adult attention was high. Smiling by children with AS evoked higher levels of adult attention, eye contact and smiling both than by chance and in comparison to other children and this effect was sustained for 30 s to 50 s. Smiling by children with AS was frequently preceded by child initiated contact toward the adult.

Discussion: The results are consistent with a kinship theory explanation of the function of heightened levels of sociability and smiling in Angelman syndrome and provide support for an emotion signalling interpretation of the mechanism by which smiling accesses social resources. Further research on other behaviours characteristic of Angelman and Prader-Willi syndromes warrant examination from this perspective.
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http://dx.doi.org/10.1111/j.1469-7610.2007.01736.xDOI Listing
June 2007

Environmental influences on the behavioral phenotype of Angelman syndrome.

Am J Ment Retard 2006 Sep;111(5):311-21

Birmingham University, UK.

Using observational methods, we examined the social influences on laughing and smiling behavior in children with Angelman syndrome by systematically manipulating aspects of social interaction. Seven boys and 4 girls who were between 4 and 11 years of age and who had a confirmed maternal deletion of chromosome 15q11-q13 completed the study. Each child was observed while repeatedly exposed to three conditions in which parameters of social interaction were manipulated. Laughing and smiling behavior varied across all children and was significantly heightened in a condition involving adult speech, touch, smiling, laughing, and eye contact. The findings highlight the importance of examining environmental and social influences on purported phenotypic behavior in genetic syndromes.
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http://dx.doi.org/10.1352/0895-8017(2006)111[311:EIOTBP]2.0.CO;2DOI Listing
September 2006