Using kinematic analyses to explore sensorimotor control impairments in children with 22q11.2 deletion syndrome.
22q11.2 deletion syndrome
ADHD
ASD
Anxiety
Coordination
Movement difficulties
Journal
Journal of neurodevelopmental disorders
ISSN: 1866-1955
Titre abrégé: J Neurodev Disord
Pays: England
ID NLM: 101483832
Informations de publication
Date de publication:
10 06 2019
10 06 2019
Historique:
received:
19
10
2018
accepted:
23
05
2019
entrez:
12
6
2019
pubmed:
12
6
2019
medline:
4
8
2020
Statut:
epublish
Résumé
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. 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. 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. 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.
Sections du résumé
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.
Identifiants
pubmed: 31182009
doi: 10.1186/s11689-019-9271-3
pii: 10.1186/s11689-019-9271-3
pmc: PMC6558818
doi:
Types de publication
Journal Article
Research Support, Non-U.S. Gov't
Langues
eng
Sous-ensembles de citation
IM
Pagination
8Subventions
Organisme : MRF
ID : MRF_MRF-154-0001-RG-SKUSE
Pays : United Kingdom
Organisme : Medical Research Council
ID : MR/P008593/1
Pays : United Kingdom
Organisme : Medical Research Council
ID : G0800509
Pays : United Kingdom
Organisme : Medical Research Council
ID : MR/L010305/1
Pays : United Kingdom
Organisme : Medical Research Council
ID : MC_PC_17210
Pays : United Kingdom
Organisme : Medical Research Council
ID : MR/L011166/1
Pays : United Kingdom
Organisme : Medical Research Council
ID : MR/K51347/1
Pays : United Kingdom
Organisme : Medical Research Council
ID : MR/N022572/1
Pays : United Kingdom
Organisme : Medical Research Council
ID : G0801418
Pays : United Kingdom
Références
Exp Brain Res. 2003 May;150(1):25-32
pubmed: 12698213
Neurosci Lett. 2006 May 22;399(3):245-8
pubmed: 16517069
Biol Psychiatry. 2006 Nov 15;60(10):1088-97
pubmed: 16806097
Mov Disord. 2006 Dec;21(12):2082-9
pubmed: 16991148
J Dev Behav Pediatr. 2007 Apr;28(2):119-24
pubmed: 17435462
Psychol Sci. 2007 Sep;18(9):796-802
pubmed: 17760776
Neurosci Lett. 2008 Aug 15;441(1):77-80
pubmed: 18597939
Child Neuropsychol. 2009 Nov;15(6):532-42
pubmed: 19280375
J Neurosci Methods. 2009 Oct 30;184(1):184-92
pubmed: 19646475
Res Dev Disabil. 2010 May-Jun;31(3):768-76
pubmed: 20181457
Res Dev Disabil. 2010 May-Jun;31(3):634-41
pubmed: 20181458
J Pediatr. 2010 Oct;157(4):658-61
pubmed: 20646714
Res Dev Disabil. 2011 Jul-Aug;32(4):1351-60
pubmed: 21334175
Neuropsychology. 2011 Nov;25(6):711-719
pubmed: 21728424
Autism. 2012 Nov;16(6):627-40
pubmed: 21949004
Res Dev Disabil. 2012 Mar-Apr;33(2):334-40
pubmed: 22119678
Res Dev Disabil. 2012 Sep-Oct;33(5):1495-502
pubmed: 22522207
Res Dev Disabil. 2013 Apr;34(4):1357-64
pubmed: 23417140
Br J Psychiatry. 2014 Jan;204(1):46-54
pubmed: 24115343
PLoS One. 2014 Feb 11;9(2):e88692
pubmed: 24523931
Am J Psychiatry. 2014 Jun;171(6):627-39
pubmed: 24577245
Exp Brain Res. 2014 Sep;232(9):2907-17
pubmed: 24825824
Biol Psychiatry. 2015 Jul 15;78(2):135-43
pubmed: 25555483
Am J Med Genet A. 2015 Mar;167A(3):639-45
pubmed: 25684639
Prenat Diagn. 2015 Aug;35(8):801-9
pubmed: 25962607
J Int Neuropsychol Soc. 2015 Jan;21(1):8-21
pubmed: 26304055
Nat Rev Dis Primers. 2015 Nov 19;1:15071
pubmed: 27189754
Mov Disord. 2016 Nov;31(11):1720-1728
pubmed: 27383763
Br J Psychiatry. 2018 Jan;212(1):27-33
pubmed: 29433607
Dev Med Child Neurol. 2019 Mar;61(3):242-285
pubmed: 30671947
Psychol Med. 1995 Jul;25(4):739-53
pubmed: 7480451