Using kinematic analyses to explore sensorimotor control impairments in children with 22q11.2 deletion syndrome / A. C. CUNNINGHAM in Journal of Neurodevelopmental Disorders, 11-1 (December 2019)  

Public ISBD [article]  inJournal of Neurodevelopmental Disorders > 11-1 (December 2019) . - 8 p. Titre : Using kinematic analyses to explore sensorimotor control impairments in children with 22q11.2 deletion syndrome Type de document : Texte imprimé et/ou numérique Auteurs : A. C. CUNNINGHAM, Auteur ; L. HILL, Auteur ; M. MON-WILLIAMS, Auteur ; K. J. PEALL, Auteur ; D. E. J. LINDEN, Auteur ; J. HALL, Auteur ; M. J. OWEN, Auteur ; M. B. M. VAN DEN BREE, Auteur Article en page(s) : 8 p. Langues : Anglais (eng) Mots-clés : 22q11.2 deletion syndrome  Adhd  Asd  Anxiety  Coordination  Movement difficulties Index. décimale : PER Périodiques 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. En ligne : https://dx.doi.org/10.1186/s11689-019-9271-3 Permalink : https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=409 [article] Using kinematic analyses to explore sensorimotor control impairments in children with 22q11.2 deletion syndrome [Texte imprimé et/ou numérique] / A. C. CUNNINGHAM, Auteur ; L. HILL, Auteur ; M. MON-WILLIAMS, Auteur ; K. J. PEALL, Auteur ; D. E. J. LINDEN, Auteur ; J. HALL, Auteur ; M. J. OWEN, Auteur ; M. B. M. VAN DEN BREE, Auteur . - 8 p. Langues : Anglais (eng) in Journal of Neurodevelopmental Disorders > 11-1 (December 2019) . - 8 p. Mots-clés : 22q11.2 deletion syndrome  Adhd  Asd  Anxiety  Coordination  Movement difficulties Index. décimale : PER Périodiques 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. En ligne : https://dx.doi.org/10.1186/s11689-019-9271-3 Permalink : https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=409

Visuospatial working memory in children and adolescents with 22q11.2 deletion syndrome; an fMRI study / R. AZUMA in Journal of Neurodevelopmental Disorders, 1-1 (March 2009)  

Public ISBD [article]  inJournal of Neurodevelopmental Disorders > 1-1 (March 2009) . - p.46-60 Titre : Visuospatial working memory in children and adolescents with 22q11.2 deletion syndrome; an fMRI study Type de document : Texte imprimé et/ou numérique Auteurs : R. AZUMA, Auteur ; Eileen DALY, Auteur ; Linda E. CAMPBELL, Auteur ; A. F. STEVENS, Auteur ; Quinton DEELEY, Auteur ; V. GIAMPIETRO, Auteur ; Michael BRAMMER, Auteur ; B. GLASER, Auteur ; F. Z. AMBERY, Auteur ; R. G. MORRIS, Auteur ; S. C. WILLIAMS, Auteur ; M. J. OWEN, Auteur ; D. G. MURPHY, Auteur ; K. C. MURPHY, Auteur Article en page(s) : p.46-60 Langues : Anglais (eng) Index. décimale : PER Périodiques Résumé : 22q11.2 deletion syndrome (22q11DS) is a genetic disorder associated with a microdeletion of chromosome 22q11. In addition to high rates of neuropsychiatric disorders such as schizophrenia and attention deficit hyperactivity disorder, children with 22q11DS have a specific neuropsychological profile with particular deficits in visuospatial and working memory. However, the neurobiological substrate underlying these deficits is poorly understood. We investigated brain function during a visuospatial working memory (SWM) task in eight children with 22q11DS and 13 healthy controls, using fMRI. Both groups showed task-related activation in dorsolateral prefrontal cortex (DLPFC) and bilateral parietal association cortices. Controls activated parietal and occipital regions significantly more than those with 22q11DS but there was no significant between-group difference in DLPFC. In addition, while controls had a significant age-related increase in the activation of posterior brain regions and an age-related decrease in anterior regions, the 22q11DS children showed the opposite pattern. Genetically determined differences in the development of specific brain systems may underpin the cognitive deficits in 22q11DS, and may contribute to the later development of neuropsychiatric disorders. En ligne : http://dx.doi.org/10.1007/s11689-009-9008-9 Permalink : https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=341 [article] Visuospatial working memory in children and adolescents with 22q11.2 deletion syndrome; an fMRI study [Texte imprimé et/ou numérique] / R. AZUMA, Auteur ; Eileen DALY, Auteur ; Linda E. CAMPBELL, Auteur ; A. F. STEVENS, Auteur ; Quinton DEELEY, Auteur ; V. GIAMPIETRO, Auteur ; Michael BRAMMER, Auteur ; B. GLASER, Auteur ; F. Z. AMBERY, Auteur ; R. G. MORRIS, Auteur ; S. C. WILLIAMS, Auteur ; M. J. OWEN, Auteur ; D. G. MURPHY, Auteur ; K. C. MURPHY, Auteur . - p.46-60. Langues : Anglais (eng) in Journal of Neurodevelopmental Disorders > 1-1 (March 2009) . - p.46-60 Index. décimale : PER Périodiques Résumé : 22q11.2 deletion syndrome (22q11DS) is a genetic disorder associated with a microdeletion of chromosome 22q11. In addition to high rates of neuropsychiatric disorders such as schizophrenia and attention deficit hyperactivity disorder, children with 22q11DS have a specific neuropsychological profile with particular deficits in visuospatial and working memory. However, the neurobiological substrate underlying these deficits is poorly understood. We investigated brain function during a visuospatial working memory (SWM) task in eight children with 22q11DS and 13 healthy controls, using fMRI. Both groups showed task-related activation in dorsolateral prefrontal cortex (DLPFC) and bilateral parietal association cortices. Controls activated parietal and occipital regions significantly more than those with 22q11DS but there was no significant between-group difference in DLPFC. In addition, while controls had a significant age-related increase in the activation of posterior brain regions and an age-related decrease in anterior regions, the 22q11DS children showed the opposite pattern. Genetically determined differences in the development of specific brain systems may underpin the cognitive deficits in 22q11DS, and may contribute to the later development of neuropsychiatric disorders. En ligne : http://dx.doi.org/10.1007/s11689-009-9008-9 Permalink : https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=341

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