Psychological well-being in parents of children with Angelman, Cornelia de Lange and Cri du Chat syndromes: Parental psychological well-being
Abstract
Background The current study focuses on mothers and fathers of children with three rare genetic syndromes that are relatively unexplored in terms of family experience: Angelman syndrome, Cornelia de Lange syndrome and Cri du Chat syndrome. Method Parents of children with Angelman syndrome (n =15), Cornelia de Lange syndrome (n = 16) and Cri du Chat syndrome (n = 18), and a matched comparison group of parents of children with autism and intellectual disabilities (n = 20) completed questionnaires on both psychological distress (stress, anxiety, depression) and positive psychological functioning. Results Parents of children with Angelman syndrome consistently reported the highest levels of psychological distress, and parents of children with Cornelia de Lange syndrome the lowest, with parents of children with Cri du Chat syndrome and autism scoring between these two. Positive psychological functioning was similar across the four aetiology groups. Conclusions Parents of children with rare genetic syndromes are at risk for high levels of stress and mental health problems. Methodological issues and the practical applications of these results are discussed.
Integrity signals
Registry
Cites
Statistics
Claims the paper makes
Claims other papers make about this one
Each is checked against this paper’s own text: does it say what the citing paper says it does?
The Positive and Negative Affect Scale positive affect subscale has previously been used with this population of parents with good reliability.
“The scale was chosen for the current study because of its briefness to administer, and previous use with this population of parents with good reliability (Cronbach's = .92; Griffith et al., 2011).”
From A mindfulness parent well-being course: evaluation of outcomes for parents of children with autism and related disabilities recruited through special schools (2018) · cites this paper as Griffith et al., 2011
Cites
Prevalence of Autism in a US Metropolitan Area
Yeargin-Allsopp, M., Rice, C., Karapurkar, T., Doernberg, N., Boyle, C., & Murphy, C. (2003) · JAMA · doi:10.1001/jama.289.1.49 · Cited by 1,248 · see citing works
Prevalence of Parent-Reported Diagnosis of Autism Spectrum Disorder Among Children in the US, 2007
Kogan, M. D., Blumberg, S. J., Schieve, L. A., Boyle, C. A., Perrin, J. M., Ghandour, R. M., Singh, G. K., Strickland, B. B., Trevathan, E., & van Dyck, P. C. (2009) · Pediatrics · doi:10.1542/peds.2009-1522 · Cited by 892 · see citing works
Normative data for the HADS from a large non-clinical sample
Crawford, J. R., Henry, J. D., Crombie, C., & Taylor, E. P. (2001) · British Journal of Clinical Psychology · doi:10.1348/014466501163904 · Cited by 815 · see citing works
Herring, S., Gray, K., Taffe, J., Tonge, B., Sweeney, D., & Einfeld, S. (2006) · Journal of Intellectual Disability Research · doi:10.1111/j.1365-2788.2006.00904.x · Cited by 588 · see citing works
Blacher, J., & McIntyre, L. L. (2006) · Journal of Intellectual Disability Research · doi:10.1111/j.1365-2788.2005.00768.x · Cited by 467 · see citing works
The Relationship Between Autism and Parenting Stress
Schieve, L. A., Blumberg, S. J., Rice, C., Visser, S. N., & Boyle, C. (2007) · Pediatrics · doi:10.1542/peds.2006-2089q · Cited by 431 · see citing works
Screening for depression in head and neck cancer
Katz, M. R., Kopek, N., Waldron, J., Devins, G. M., & Tomlinson, G. (2004) · Psycho-Oncology · doi:10.1002/pon.734 · Cited by 259 · see citing works
MacDonald, E. E., Hastings, R. P., & Fitzsimons, E. (2010) · Journal of Applied Research in Intellectual Disabilities · doi:10.1111/j.1468-3148.2009.00546.x · Cited by 101 · see citing works
Self-citation
Flagged first, then by citations
Cited by
Sorted by citations
Jones, L., Hastings, R. P., Totsika, V., Keane, L., & Rhule, N. (2014) · American Journal on Intellectual and Developmental Disabilities · doi:10.1352/1944-7558-119.2.171 · Cited by 192 · see citing works
Self-citationJones, L., Gold, E., Totsika, V., Hastings, R. P., Jones, M., Griffiths, A., & Silverton, S. (2018) · European Journal of Special Needs Education · doi:10.1080/08856257.2017.1297571 · Cited by 68 · see citing works
Self-citationAdams, D., Clarke, S., Griffith, G., Howlin, P., Moss, J., Petty, J., Tunnicliffe, P., & Oliver, C. (2018) · American Journal on Intellectual and Developmental Disabilities · doi:10.1352/1944-7558-123.3.241 · Cited by 31 · see citing works
Self-citationCacioppo, C. N., Conway, L. J., Mehta, D., Krantz, I. D., & Noon, S. E. (2016) · American Journal of Medical Genetics Part C: Seminars in Medical Genetics · doi:10.1002/ajmg.c.31504 · Cited by 14 · see citing works
Chilvers, R., Gratton, S., & Bernard, S. H. (2013) · Advances in Mental Health and Intellectual Disabilities · doi:10.1108/20441281311294701 · Cited by 8 · see citing works
- Authors
- Griffith, G. M., Hastings, R. P., Oliver, C., Howlin, P., Moss, J., Petty, J., & Tunnicliffe, P.
- Affiliations
- Bangor University (GB)
- University of Birmingham (GB)
- Corresponding author
- Griffith, G. M.
- Venue
- Journal of Intellectual Disability Research
- Publisher
- Wiley
- Published
- 2011-02-15
- Volume
- 55
- Issue
- 4
- Pages
- 397-410
- ISSN
- 0964-2633 (ISSN-L)
- OpenAlex
- W1499900727
- Cited by
- 88
- References
- 8
- Citations by year
- 2026: 2 · 2025: 6 · 2024: 5 · 2023: 6 · 2022: 4 · 2021: 2 · 2020: 8 · 2019: 7 · 2018: 8 · 2017: 11 · 2016: 7 · 2015: 2 · 2014: 6 · 2013: 8 · 2012: 3
- FWCI
- 6.90
- Type
- journal article
- Language
- en
- Open access
- closed
- Locations
- Journal of Intellectual Disability Research (publishedVersion)
- PubMed (publishedVersion)
- UCL Discovery (University College London) (submittedVersion)
- Research Portal (King's College London) (submittedVersion)
- University of Birmingham Research Portal (University of Birmingham) (submittedVersion)
- Topics
- Social Sciences › Psychology › Clinical Psychology › Family and Disability Support Research
- Life Sciences › Biochemistry, Genetics and Molecular Biology › Genetics › Genomics and Rare Diseases
- Health Sciences › Medicine › Public Health, Environmental and Occupational Health › Down syndrome and intellectual disability research
- Keywords
- Angelman syndrome, Cornelia de Lange syndrome, cri du chat syndrome, rare genetic disorders, parental psychological well-being, psychological distress, parental stress, anxiety, depression, positive psychological functioning, parents of children with disabilities, autism spectrum disorder, intellectual disability
- MeSH terms
- Adaptation, Psychological, Adolescent, Analysis of Variance, Anxiety, Autistic Disorder, Child, Child Behavior Disorders, Developmental Disabilities, Child Rearing, Child, Preschool, Cri-du-Chat Syndrome, De Lange Syndrome, Depression, Female, Humans, Male, Parent-Child Relations, Parents, Sex Factors, Social Support, Stress, Psychological, Case-Control Studies, Self-Injurious Behavior, Caregivers, Angelman Syndrome, Chromosome Disorders, Young Adult
About these checks
Registry, retraction, Cites and statistics results are computed, not judged, with one exception: a printed reference that no index lists is read once by a model before it counts as a citation error. Claims are extracted by a model and kept only when their quote appears verbatim in the text; each cited source is then checked, with a model’s help, for whether it says what the claim relies on. Those verdicts can be wrong — read the claim’s quote and the cited source before relying on one.
Statistics and arithmetic are recomputed, not judged.
Confidence bands are shown only for fields calibrated on our annotated test set. None is calibrated yet, so no band is shown.
What counts as a claim ›How we measure confidence ›How we check this ›
Are you an author, or do you see a problem with this page? Request a review
