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Using kinematic analyses to explore sensorimotor control impairments in children with 22q11.2 deletion syndrome

BACKGROUND: The 22q11.2 deletion is associated with psychiatric and behavioural disorders, intellectual disability and multiple physical abnormalities. Recent research also indicates impaired coordination skills may be part of the clinical phenotype. This study aimed to characterise sensorimotor con...

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Autores principales: Cunningham, Adam C., Hill, Liam, Mon-Williams, Mark, Peall, Kathryn J., Linden, David E. J., Hall, Jeremy, Owen, Michael J., van den Bree, Marianne B. M.
Formato: Online Artículo Texto
Lenguaje:English
Publicado: BioMed Central 2019
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC6558818/
https://www.ncbi.nlm.nih.gov/pubmed/31182009
http://dx.doi.org/10.1186/s11689-019-9271-3
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author Cunningham, Adam C.
Hill, Liam
Mon-Williams, Mark
Peall, Kathryn J.
Linden, David E. J.
Hall, Jeremy
Owen, Michael J.
van den Bree, Marianne B. M.
author_facet Cunningham, Adam C.
Hill, Liam
Mon-Williams, Mark
Peall, Kathryn J.
Linden, David E. J.
Hall, Jeremy
Owen, Michael J.
van den Bree, Marianne B. M.
author_sort Cunningham, Adam C.
collection PubMed
description BACKGROUND: The 22q11.2 deletion is associated with psychiatric and behavioural disorders, intellectual disability and multiple physical abnormalities. Recent research also indicates impaired coordination skills may be part of the clinical phenotype. This study aimed to characterise sensorimotor control abilities in children with 22q11.2 deletion syndrome (22q11.2DS) and investigate their relationships with co-occurring IQ impairments and psychopathology. METHODS: Fifty-four children with 22q11.2DS and 24 unaffected sibling controls, comparable in age and gender, underwent kinematic analysis of their hand movements, whilst performing a battery of three visuo-manual coordination tasks that measured their tracking, aiming and steering abilities. Additionally, standardised assessments of full-scale IQ (FSIQ), attention deficit hyperactivity disorder, indicative autism spectrum disorder (ASD) and anxiety disorder symptomatology were conducted. RESULTS: Children with 22q11.2DS showed deficits on seven of eight kinematic descriptors of movement quality across the three coordination tasks, compared to controls. Within 22q11.2DS cases, the extent of impairment on only three kinematic descriptors was significantly related to FSIQ after correction for multiple testing. Moreover, only error whilst visuo-manually tracking was nominally associated with ADHD symptom counts. CONCLUSIONS: Impairments in sensorimotor control are seen on a range of visuo-manual tasks in children with 22q11.2DS but the extent of these impairments are largely unrelated to the severity of other psychopathological and intellectual impairments commonly found in children with 22q11.2DS. ELECTRONIC SUPPLEMENTARY MATERIAL: The online version of this article (10.1186/s11689-019-9271-3) contains supplementary material, which is available to authorized users.
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spelling pubmed-65588182019-06-13 Using kinematic analyses to explore sensorimotor control impairments in children with 22q11.2 deletion syndrome Cunningham, Adam C. Hill, Liam Mon-Williams, Mark Peall, Kathryn J. Linden, David E. J. Hall, Jeremy Owen, Michael J. van den Bree, Marianne B. M. J Neurodev Disord Research BACKGROUND: The 22q11.2 deletion is associated with psychiatric and behavioural disorders, intellectual disability and multiple physical abnormalities. Recent research also indicates impaired coordination skills may be part of the clinical phenotype. This study aimed to characterise sensorimotor control abilities in children with 22q11.2 deletion syndrome (22q11.2DS) and investigate their relationships with co-occurring IQ impairments and psychopathology. METHODS: Fifty-four children with 22q11.2DS and 24 unaffected sibling controls, comparable in age and gender, underwent kinematic analysis of their hand movements, whilst performing a battery of three visuo-manual coordination tasks that measured their tracking, aiming and steering abilities. Additionally, standardised assessments of full-scale IQ (FSIQ), attention deficit hyperactivity disorder, indicative autism spectrum disorder (ASD) and anxiety disorder symptomatology were conducted. RESULTS: Children with 22q11.2DS showed deficits on seven of eight kinematic descriptors of movement quality across the three coordination tasks, compared to controls. Within 22q11.2DS cases, the extent of impairment on only three kinematic descriptors was significantly related to FSIQ after correction for multiple testing. Moreover, only error whilst visuo-manually tracking was nominally associated with ADHD symptom counts. CONCLUSIONS: Impairments in sensorimotor control are seen on a range of visuo-manual tasks in children with 22q11.2DS but the extent of these impairments are largely unrelated to the severity of other psychopathological and intellectual impairments commonly found in children with 22q11.2DS. ELECTRONIC SUPPLEMENTARY MATERIAL: The online version of this article (10.1186/s11689-019-9271-3) contains supplementary material, which is available to authorized users. BioMed Central 2019-06-10 /pmc/articles/PMC6558818/ /pubmed/31182009 http://dx.doi.org/10.1186/s11689-019-9271-3 Text en © The Author(s). 2019 Open AccessThis article is distributed under the terms of the Creative Commons Attribution 4.0 International License (http://creativecommons.org/licenses/by/4.0/), which permits unrestricted use, distribution, and reproduction in any medium, provided you give appropriate credit to the original author(s) and the source, provide a link to the Creative Commons license, and indicate if changes were made. The Creative Commons Public Domain Dedication waiver (http://creativecommons.org/publicdomain/zero/1.0/) applies to the data made available in this article, unless otherwise stated.
spellingShingle Research
Cunningham, Adam C.
Hill, Liam
Mon-Williams, Mark
Peall, Kathryn J.
Linden, David E. J.
Hall, Jeremy
Owen, Michael J.
van den Bree, Marianne B. M.
Using kinematic analyses to explore sensorimotor control impairments in children with 22q11.2 deletion syndrome
title Using kinematic analyses to explore sensorimotor control impairments in children with 22q11.2 deletion syndrome
title_full Using kinematic analyses to explore sensorimotor control impairments in children with 22q11.2 deletion syndrome
title_fullStr Using kinematic analyses to explore sensorimotor control impairments in children with 22q11.2 deletion syndrome
title_full_unstemmed Using kinematic analyses to explore sensorimotor control impairments in children with 22q11.2 deletion syndrome
title_short Using kinematic analyses to explore sensorimotor control impairments in children with 22q11.2 deletion syndrome
title_sort using kinematic analyses to explore sensorimotor control impairments in children with 22q11.2 deletion syndrome
topic Research
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC6558818/
https://www.ncbi.nlm.nih.gov/pubmed/31182009
http://dx.doi.org/10.1186/s11689-019-9271-3
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