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Cited by 31Family and Disability Support ResearchChild and Adolescent Psychosocial and Emotional DevelopmentGenomics and Rare Diseases

Mental Health and Well-Being in Mothers of Children With Rare Genetic Syndromes Showing Chronic Challenging Behavior: A Cross-Sectional and Longitudinal Study

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Adams, Dawn, Clarke, Samantha, Griffith, Gemma, Howlin, Pat, Moss, Jo, Petty, Jane, Tunnicliffe, Penny, Oliver, Chris·2018·American Journal on Intellectual and Developmental Disabilities
DOI 10.1352/1944-7558-123.3.241
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Abstract

It is well documented that mothers of children with challenging behavior (CB) experience elevated levels of stress and that this persists over time, but less is known about the experience of mothers of children with rare genetic syndromes. This article describes 2 studies, 1 cross-sectional and 1 longitudinal, comparing well-being in mothers of children with Angelman, Cornelia de Lange and Cri du Chat syndrome who have either shown chronic CB (n = 18) or low/no CB (n = 26) in the preceding 7 years. The presence of chronic, long-term CB increased maternal stress but not depression or anxiety, and did not influence positive well-being. Stress relating specifically to their child's genetic syndrome reduced with age, highlighting the need for further exploration in this area.

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  • The quality of life of patients with ROD is lower than for the general population in three key areas: physical limitations, social isolation and stigmatization, and emotional distress.

    “The quality of life of patients with ROD is lower than for the general population in three key areas: physical limitations (movements, pain, sleep problems, attention or concentration problems that affect daily life), [3–9], social isolation and stigmatization [3, 7, 9–13], and emotional distress (anger, depression, anxiety, hopelessness, loss, lack of control, abandonment) [3, 5, 8, 14–17].”

    From Connect-ROD – development and qualitative evaluation of a community-based group intervention to support well-being in patients with a rare or orphan disease (2024) · cites this paper as [4]

    The Introduction and Discussion examine stress, anxiety, and depression in mothers of children with rare genetic syndromes, not quality of life in the patients themselves. Searches for patient quality of life, physical limitations, social isolation, and stigmatization found no passage supporting the claimed comparison with the general population. (LLM, conf=0.76)

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Research works printed but not linked: 3

  1. Pit-ten Cate, I. (2003). Positive gain in mothers of children with physical disabilities. Unpublished PhD Thesis. University of Southampton, England, UK.
  2. Seltzer, M. M., & Greenberg, J. S. (2001). Life course impacts of parenting a child with a disability. American Journal of Mental Retardation, 106(3), 265-286.
  3. Sparrow, S., Cichetti, D., & Balla, D.A. (2005). Vineland Adaptive Behavior Scales 2. Minneapolis, MN: NCS Pearson, Inc; 2005.

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Authors
Adams, D., Clarke, S., Griffith, G., Howlin, P., Moss, J., Petty, J., Tunnicliffe, P., & Oliver, C.
Affiliations
  • Griffith University (AU)
  • Bangor University (GB)
  • Pat Howlin, Kings College, London, UK;
  • Jo Moss, University of Birmingham, UK;
  • King's College London (GB)
  • University of Birmingham (GB)
Corresponding author
Adams, Dawn
ORCID
Venue
American Journal on Intellectual and Developmental Disabilities
Publisher
Informa UK Limited
Published
2018-05-01
Volume
123
Issue
3
Pages
241-253
ISSN
  • 1944-7558 (ISSN-L)
  • 1944-7515
DOI
10.1352/1944-7558-123.3.241
OpenAlex
W2803017423
Cited by
31
References
38
Citations by year
2026: 2 · 2025: 5 · 2024: 5 · 2023: 4 · 2022: 4 · 2021: 2 · 2020: 3 · 2019: 4 · 2018: 1 · 2014: 1
FWCI
2.93
Type
journal article
Language
en
Open access
Locations
  • American Journal on Intellectual and Developmental Disabilities (publishedVersion)
  • PubMed (publishedVersion)
  • University of Birmingham Research Portal (University of Birmingham) (submittedVersion, other-oa)
  • Griffith Research Online (submittedVersion)
Funders
  • National Institute for Health and Care Research (PDF-2010-03-02)
  • Cerebra
Topics
  • Social Sciences › Psychology › Clinical Psychology › Family and Disability Support Research
  • Social Sciences › Psychology › Clinical Psychology › Child and Adolescent Psychosocial and Emotional Development
  • Life Sciences › Biochemistry, Genetics and Molecular Biology › Genetics › Genomics and Rare Diseases
Keywords
maternal mental health, maternal well-being, behavioral problems, Angelman syndrome, Cornelia de Lange syndrome, cri du chat syndrome, maternal stress, depression, anxiety
MeSH terms
Problem Behavior, Activities of Daily Living, Adolescent, Adult, Anxiety, Child, Cri-du-Chat Syndrome, Cross-Sectional Studies, De Lange Syndrome, Depression, Female, Humans, Longitudinal Studies, Male, Mental Health, Middle Aged, Mothers, Stress, Psychological, Case-Control Studies, Angelman Syndrome, Rare Diseases, Young Adult

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