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Child characteristics associated with child quality of life and parenting stress in Angelman syndrome.

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Title: Child characteristics associated with child quality of life and parenting stress in Angelman syndrome.
Authors: Hagenaar, D. A. (AUTHOR), Bindels‐de Heus, K. G. C. B. (AUTHOR), Lubbers, K. (AUTHOR), ten Hoopen, L. W. (AUTHOR), Rietman, A. B. (AUTHOR), de Nijs, P. F. A. (AUTHOR), Hillegers, M. H. J. (AUTHOR), Moll, H. A. (AUTHOR), de Wit, M. C. Y. (AUTHOR), Dieleman, G. C. (AUTHOR), Mous, S. E. (AUTHOR)
Source: Journal of Intellectual Disability Research. Mar2024, Vol. 68 Issue 3, p248-263. 16p.
Subjects: Research funding, Angelman syndrome, Mental illness, Questionnaires, Hospitals, Affective disorders, Age distribution, Longitudinal method, Behavior disorders in children, Quality of life, Psychological stress, Child Behavior Checklist, Parents of children with disabilities, Psychosocial factors, Regression analysis, Genotypes, Sleep disorders, Disease complications, Symptoms, Children
Geographic Terms: Netherlands
Abstract: Background: Angelman syndrome (AS) is a rare neurodevelopmental disorder characterised by severe intellectual disability, movement disorder, epilepsy, sleeping problems, and behavioural issues. Little is known on child health‐related quality of life (HRQoL) in AS. AS family studies have reported elevated parenting stress and a high impact of the child's syndrome on the parent. It is unclear which factors influence child HRQoL and parenting stress/impact in AS. Methods: We collected data prospectively through standardised clinical assessments of children with AS at the ENCORE Expertise centre for Angelman Syndrome at the Erasmus MC Sophia Children's Hospital. A linear regression analysis was conducted for the following outcome variables: (1) child HRQoL (Infant and Toddler Quality of Life Questionnaire); (2) the impact of the child's syndrome on the parent (Infant and Toddler Quality of Life Questionnaire); and (3) parenting stress (Parenting Stress Index). Predictor variables were child genotype, epilepsy, sleeping problems (Sleep Disturbance Scale for Children), cognitive developmental level (Bayley Cognition Scale), autistic features (Autism Diagnostic Observation Schedule) and emotional/behavioural problems (Child Behaviour Checklist). Covariates were sex, age and socio‐economic status. Results: The study sample consisted of 73 children with AS, mean age = 9.1 years, range = 2–18 years. Emotional/behavioural problems were the strongest significant predictor of lowered child HRQoL. Internalising problems were driving this effect. In addition, having the deletion genotype and higher age was related to lower child HRQoL. Sleeping problems were related to a higher impact of the child's syndrome on the parent. Finally, emotional/behavioural problems were associated with higher parenting stress. Cognitive developmental level, autistic features and epilepsy were not a significant predictor of child HRQoL and parenting stress/impact. Conclusions: These results suggest that interventions aimed at increasing child HRQoL and decreasing parenting stress/impact in AS should focus on child emotional/behavioural problems and sleeping problems, using a family‐centred approach. [ABSTRACT FROM AUTHOR]
Copyright of Journal of Intellectual Disability Research is the property of Wiley-Blackwell and its content may not be copied or emailed to multiple sites without the copyright holder's express written permission. Additionally, content may not be used with any artificial intelligence tools or machine learning technologies. However, users may print, download, or email articles for individual use. This abstract may be abridged. No warranty is given about the accuracy of the copy. Users should refer to the original published version of the material for the full abstract. (Copyright applies to all Abstracts.)
Database: Psychology and Behavioral Sciences Collection
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  Data: Child characteristics associated with child quality of life and parenting stress in Angelman syndrome.
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  Data: <searchLink fieldCode="AR" term="%22Hagenaar%2C+D%2E+A%2E%22">Hagenaar, D. A.</searchLink> (AUTHOR)<br /><searchLink fieldCode="AR" term="%22Bindels‐de+Heus%2C+K%2E+G%2E+C%2E+B%2E%22">Bindels‐de Heus, K. G. C. B.</searchLink> (AUTHOR)<br /><searchLink fieldCode="AR" term="%22Lubbers%2C+K%2E%22">Lubbers, K.</searchLink> (AUTHOR)<br /><searchLink fieldCode="AR" term="%22ten+Hoopen%2C+L%2E+W%2E%22">ten Hoopen, L. W.</searchLink> (AUTHOR)<br /><searchLink fieldCode="AR" term="%22Rietman%2C+A%2E+B%2E%22">Rietman, A. B.</searchLink> (AUTHOR)<br /><searchLink fieldCode="AR" term="%22de+Nijs%2C+P%2E+F%2E+A%2E%22">de Nijs, P. F. A.</searchLink> (AUTHOR)<br /><searchLink fieldCode="AR" term="%22Hillegers%2C+M%2E+H%2E+J%2E%22">Hillegers, M. H. J.</searchLink> (AUTHOR)<br /><searchLink fieldCode="AR" term="%22Moll%2C+H%2E+A%2E%22">Moll, H. A.</searchLink> (AUTHOR)<br /><searchLink fieldCode="AR" term="%22de+Wit%2C+M%2E+C%2E+Y%2E%22">de Wit, M. C. Y.</searchLink> (AUTHOR)<br /><searchLink fieldCode="AR" term="%22Dieleman%2C+G%2E+C%2E%22">Dieleman, G. C.</searchLink> (AUTHOR)<br /><searchLink fieldCode="AR" term="%22Mous%2C+S%2E+E%2E%22">Mous, S. E.</searchLink> (AUTHOR)
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  Data: <searchLink fieldCode="JN" term="%22Journal+of+Intellectual+Disability+Research%22">Journal of Intellectual Disability Research</searchLink>. Mar2024, Vol. 68 Issue 3, p248-263. 16p.
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  Data: <searchLink fieldCode="DE" term="%22Research+funding%22">Research funding</searchLink><br /><searchLink fieldCode="DE" term="%22Angelman+syndrome%22">Angelman syndrome</searchLink><br /><searchLink fieldCode="DE" term="%22Mental+illness%22">Mental illness</searchLink><br /><searchLink fieldCode="DE" term="%22Questionnaires%22">Questionnaires</searchLink><br /><searchLink fieldCode="DE" term="%22Hospitals%22">Hospitals</searchLink><br /><searchLink fieldCode="DE" term="%22Affective+disorders%22">Affective disorders</searchLink><br /><searchLink fieldCode="DE" term="%22Age+distribution%22">Age distribution</searchLink><br /><searchLink fieldCode="DE" term="%22Longitudinal+method%22">Longitudinal method</searchLink><br /><searchLink fieldCode="DE" term="%22Behavior+disorders+in+children%22">Behavior disorders in children</searchLink><br /><searchLink fieldCode="DE" term="%22Quality+of+life%22">Quality of life</searchLink><br /><searchLink fieldCode="DE" term="%22Psychological+stress%22">Psychological stress</searchLink><br /><searchLink fieldCode="DE" term="%22Child+Behavior+Checklist%22">Child Behavior Checklist</searchLink><br /><searchLink fieldCode="DE" term="%22Parents+of+children+with+disabilities%22">Parents of children with disabilities</searchLink><br /><searchLink fieldCode="DE" term="%22Psychosocial+factors%22">Psychosocial factors</searchLink><br /><searchLink fieldCode="DE" term="%22Regression+analysis%22">Regression analysis</searchLink><br /><searchLink fieldCode="DE" term="%22Genotypes%22">Genotypes</searchLink><br /><searchLink fieldCode="DE" term="%22Sleep+disorders%22">Sleep disorders</searchLink><br /><searchLink fieldCode="DE" term="%22Disease+complications%22">Disease complications</searchLink><br /><searchLink fieldCode="DE" term="%22Symptoms%22">Symptoms</searchLink><br /><searchLink fieldCode="DE" term="%22Children%22">Children</searchLink>
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  Data: <searchLink fieldCode="DE" term="%22Netherlands%22">Netherlands</searchLink>
– Name: Abstract
  Label: Abstract
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  Data: Background: Angelman syndrome (AS) is a rare neurodevelopmental disorder characterised by severe intellectual disability, movement disorder, epilepsy, sleeping problems, and behavioural issues. Little is known on child health‐related quality of life (HRQoL) in AS. AS family studies have reported elevated parenting stress and a high impact of the child's syndrome on the parent. It is unclear which factors influence child HRQoL and parenting stress/impact in AS. Methods: We collected data prospectively through standardised clinical assessments of children with AS at the ENCORE Expertise centre for Angelman Syndrome at the Erasmus MC Sophia Children's Hospital. A linear regression analysis was conducted for the following outcome variables: (1) child HRQoL (Infant and Toddler Quality of Life Questionnaire); (2) the impact of the child's syndrome on the parent (Infant and Toddler Quality of Life Questionnaire); and (3) parenting stress (Parenting Stress Index). Predictor variables were child genotype, epilepsy, sleeping problems (Sleep Disturbance Scale for Children), cognitive developmental level (Bayley Cognition Scale), autistic features (Autism Diagnostic Observation Schedule) and emotional/behavioural problems (Child Behaviour Checklist). Covariates were sex, age and socio‐economic status. Results: The study sample consisted of 73 children with AS, mean age = 9.1 years, range = 2–18 years. Emotional/behavioural problems were the strongest significant predictor of lowered child HRQoL. Internalising problems were driving this effect. In addition, having the deletion genotype and higher age was related to lower child HRQoL. Sleeping problems were related to a higher impact of the child's syndrome on the parent. Finally, emotional/behavioural problems were associated with higher parenting stress. Cognitive developmental level, autistic features and epilepsy were not a significant predictor of child HRQoL and parenting stress/impact. Conclusions: These results suggest that interventions aimed at increasing child HRQoL and decreasing parenting stress/impact in AS should focus on child emotional/behavioural problems and sleeping problems, using a family‐centred approach. [ABSTRACT FROM AUTHOR]
– Name: AbstractSuppliedCopyright
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  Data: <i>Copyright of Journal of Intellectual Disability Research is the property of Wiley-Blackwell and its content may not be copied or emailed to multiple sites without the copyright holder's express written permission. Additionally, content may not be used with any artificial intelligence tools or machine learning technologies. However, users may print, download, or email articles for individual use. This abstract may be abridged. No warranty is given about the accuracy of the copy. Users should refer to the original published version of the material for the full abstract.</i> (Copyright applies to all Abstracts.)
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RecordInfo BibRecord:
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    Identifiers:
      – Type: doi
        Value: 10.1111/jir.13106
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      – Code: eng
        Text: English
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        PageCount: 16
        StartPage: 248
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      – SubjectFull: Research funding
        Type: general
      – SubjectFull: Angelman syndrome
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      – SubjectFull: Mental illness
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      – SubjectFull: Questionnaires
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      – SubjectFull: Hospitals
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      – SubjectFull: Affective disorders
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      – SubjectFull: Age distribution
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      – SubjectFull: Longitudinal method
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      – SubjectFull: Behavior disorders in children
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      – SubjectFull: Quality of life
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      – SubjectFull: Genotypes
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      – SubjectFull: Netherlands
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