The behavioural phenotype of Smith-Magenis syndrome: evidence for a gene-environment interaction

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The behavioural phenotype of Smith-Magenis syndrome: evidence for a gene-environment interaction. / Taylor, L; Oliver, Christopher.

In: Journal of Autism and Developmental Disorders, Vol. 52, No. 10, 01.10.2008, p. 830-41.

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@article{e96f187941354d2181bee420225da563,
title = "The behavioural phenotype of Smith-Magenis syndrome: evidence for a gene-environment interaction",
abstract = "BACKGROUND: Behaviour problems and a preference for adult contact are reported to be prominent in the phenotype of Smith-Magenis syndrome. In this study we examined the relationship between social interactions and self-injurious and aggressive/disruptive behaviour in Smith-Magenis syndrome to explore potential operant reinforcement of problem behaviours and thus a gene-environment interaction. METHOD: Observational data on five children with Smith-Magenis syndrome (age range 3 to 13 years) were collected for between 9 and 12 h. The associations between purported phenotypic behaviours and two environmental events (adult attention and demands) were examined using descriptive analysis. RESULTS: All participants engaged in self-injurious behaviour and aggressive/disruptive outbursts. Sequential analyses of aggressive/disruptive outbursts and self-injury revealed that these behaviours were evoked by low levels of adult attention and led to increased levels of attention following the behaviours in three and two participants respectively out of the four for whom this analysis was possible. CONCLUSIONS: Problem behaviour in Smith-Magenis syndrome was evoked by decreased social contact in three out of four children. These data, considered alongside the preference for adult contact and the significantly increased prevalence of these behaviours in Smith-Magenis syndrome, illustrate a potential gene-environment interaction for problem behaviour in this syndrome.",
keywords = "aggression, behavioural phenotype, self-injurious behaviour, functional analysis, Smith-Magenis syndrome",
author = "L Taylor and Christopher Oliver",
year = "2008",
month = oct,
day = "1",
doi = "10.1111/j.1365-2788.2008.01066.x",
language = "English",
volume = "52",
pages = "830--41",
journal = "Journal of Autism and Developmental Disorders",
issn = "0162-3257",
publisher = "Springer",
number = "10",

}

RIS

TY - JOUR

T1 - The behavioural phenotype of Smith-Magenis syndrome: evidence for a gene-environment interaction

AU - Taylor, L

AU - Oliver, Christopher

PY - 2008/10/1

Y1 - 2008/10/1

N2 - BACKGROUND: Behaviour problems and a preference for adult contact are reported to be prominent in the phenotype of Smith-Magenis syndrome. In this study we examined the relationship between social interactions and self-injurious and aggressive/disruptive behaviour in Smith-Magenis syndrome to explore potential operant reinforcement of problem behaviours and thus a gene-environment interaction. METHOD: Observational data on five children with Smith-Magenis syndrome (age range 3 to 13 years) were collected for between 9 and 12 h. The associations between purported phenotypic behaviours and two environmental events (adult attention and demands) were examined using descriptive analysis. RESULTS: All participants engaged in self-injurious behaviour and aggressive/disruptive outbursts. Sequential analyses of aggressive/disruptive outbursts and self-injury revealed that these behaviours were evoked by low levels of adult attention and led to increased levels of attention following the behaviours in three and two participants respectively out of the four for whom this analysis was possible. CONCLUSIONS: Problem behaviour in Smith-Magenis syndrome was evoked by decreased social contact in three out of four children. These data, considered alongside the preference for adult contact and the significantly increased prevalence of these behaviours in Smith-Magenis syndrome, illustrate a potential gene-environment interaction for problem behaviour in this syndrome.

AB - BACKGROUND: Behaviour problems and a preference for adult contact are reported to be prominent in the phenotype of Smith-Magenis syndrome. In this study we examined the relationship between social interactions and self-injurious and aggressive/disruptive behaviour in Smith-Magenis syndrome to explore potential operant reinforcement of problem behaviours and thus a gene-environment interaction. METHOD: Observational data on five children with Smith-Magenis syndrome (age range 3 to 13 years) were collected for between 9 and 12 h. The associations between purported phenotypic behaviours and two environmental events (adult attention and demands) were examined using descriptive analysis. RESULTS: All participants engaged in self-injurious behaviour and aggressive/disruptive outbursts. Sequential analyses of aggressive/disruptive outbursts and self-injury revealed that these behaviours were evoked by low levels of adult attention and led to increased levels of attention following the behaviours in three and two participants respectively out of the four for whom this analysis was possible. CONCLUSIONS: Problem behaviour in Smith-Magenis syndrome was evoked by decreased social contact in three out of four children. These data, considered alongside the preference for adult contact and the significantly increased prevalence of these behaviours in Smith-Magenis syndrome, illustrate a potential gene-environment interaction for problem behaviour in this syndrome.

KW - aggression

KW - behavioural phenotype

KW - self-injurious behaviour

KW - functional analysis

KW - Smith-Magenis syndrome

U2 - 10.1111/j.1365-2788.2008.01066.x

DO - 10.1111/j.1365-2788.2008.01066.x

M3 - Article

C2 - 18466291

VL - 52

SP - 830

EP - 841

JO - Journal of Autism and Developmental Disorders

JF - Journal of Autism and Developmental Disorders

SN - 0162-3257

IS - 10

ER -