ArticleAmerican journal of medical genetics. Part A2019
Maladaptive behaviors in individuals with Angelman syndrome.
Article in American journal of medical genetics. Part A, 2019. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 22 papers, 2 of them syntheses that pooled it.
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Who cites it
22 citing papers in PubMed, 2 syntheses or guidelines pooled it, 47 citations in OpenAlex.
- The Impact of Fragile X Syndrome on Caregivers: A Systematic Review.Journal of intellectual disability research : JIDR · 2026Pooled it
- Quality of life of parents with children with congenital abnormalities: a systematic review with meta-analysis of assessment methods and levels of quality of life.Quality of life research : an international journal of quality of life aspects of treatment, care and rehabilitation · 2022Pooled it
- Development of at-home video recordings for functional skill assessment in Angelman Syndrome: a pilot study.Journal of neurodevelopmental disorders · 2026Article
- Criterion Validity, Scalability and Stability of Scoring on the Bayley-III in Children With Angelman Syndrome.Journal of intellectual disability research : JIDR · 2025Article
- Behavioral and molecular insights into anxiety in ube3a and fmr1 zebrafish models of autism spectrum disorders.Translational psychiatry · 2025Article
- Longitudinal characterization of clinical, developmental, and behavioral phenotypes in 101 children and adults with FOXG1 syndrome.Journal of neurodevelopmental disorders · 2025Article
- Parental Stress and Family Quality of Life in Families of Individuals Living With Angelman Syndrome.Journal of intellectual disability research : JIDR · 2025Article
- Symptom network analysis of pregnancy stress, depressive symptoms and quality of life: a cross-sectional study of pregnant women in Xinjiang, China, 2023-2024.BMC psychology · 2025Article
- Using the Bayley-4 and Vineland-3 in Angelman syndrome: barriers, solutions, and challenging items.Orphanet journal of rare diseases · 2025Article
- Perception of psychosocial burden in mothers of children with rare pediatric neurological diseases.Scientific reports · 2025Article
- Enabling endpoint development for interventional clinical trials in individuals with Angelman syndrome: a prospective, longitudinal, observational clinical study (FREESIAS).Journal of neurodevelopmental disorders · 2023Observational
- [Angelman syndrome in adulthood].Revista de neurologia · 2023Article
- Social attention and social-emotional modulation of attention in Angelman syndrome: an eye-tracking study.Scientific reports · 2023Article
- Association between early and current gastro-intestinal symptoms and co-morbidities in children and adolescents with Angelman syndrome.Journal of intellectual disability research : JIDR · 2022Article
- Methylation analysis and developmental profile of two individuals with Angelman syndrome due to mosaic imprinting defects.European journal of medical genetics · 2022Article
- A multidisciplinary approach and consensus statement to establish standards of care for Angelman syndrome.Molecular genetics & genomic medicine · 2022Review
- Parenting Stress in Mothers of Children and Adolescents with Down Syndrome.Journal of clinical medicine · 2022Article
- Anxiety in Angelman Syndrome.American journal on intellectual and developmental disabilities · 2022Article
- Dissociating motor impairment from five-choice serial reaction time task performance in a mouse model of Angelman syndrome.Frontiers in behavioral neuroscience · 2022Article
- Clinical aspects of a large group of adults with Angelman syndrome.American journal of medical genetics. Part A · 2021Article
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Authors and funding
12 authors at 7 institutions in 2 countries.
Funding
Abstract
Maladaptive behaviors are challenging and a source of stress for caregivers of individuals with Angelman Syndrome (AS). There is limited information on how these maladaptive behaviors vary over time among individuals with AS due to different genetic etiologies. In this study, caregivers of 301 individuals with AS were asked questions about their child's behavior and completed the Aberrant Behavior Checklist-Community version (ABC-C). Developmental functioning was evaluated with either the Bayley Scales of Infant Development, Third Edition (Bayley-III) or the Mullen Scales of Early Learning (MSEL). Family functioning was assessed using the parent-completed Parenting Stress Index (PSI) and the Family Quality of Life questionnaire (FQoL). Approximately 70% of participants had AS due to a deletion on the maternally-inherited copy of chromosome 15q11q13. Results revealed that at baseline, individuals with AS had low scores in the domains of lethargy (mean: 2.6-4.2 depending on genotype) and stereotypy (mean: 2.3-4.2 depending on genotype). Higher cognitive functioning was associated with increased irritability (r = 0.32, p < .01). Hyperactivity (p < .05) and irritability (p < .05) increased with age across all genotypes and should be ongoing targets for both behavioral and pharmacological treatment. Concerns for short attention span were endorsed by more than 70% of caregivers at baseline. Maladaptive behaviors, particularly hyperactivity, irritability and aggression, adversely affected parental stress, and family quality of life.
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