Perceived quality of life among caregivers of children with a childhood-onset dystrophinopathy: a double ABCX model of caregiver stressors and perceived resources

Details

• Alternative Title:
  Health Qual Life Outcomes
• Personal Author:
  Frishman, Natalia ; Conway, Kristin Caspers ; Andrews, Jennifer ; Oleson, Jacob ; Mathews, Katherine ; Ciafaloni, Emma ; Oleszek, Joyce ; Lamb, Molly ; Matthews, Dennis ; Paramsothy, Pangaja ; McKirgan, Lowell ; Romitti, Paul
• Description:
  Background
  
  Duchenne and Becker muscular dystrophies, collectively referred to as dystrophinopathies, are recessive X-linked disorders characterized by progressive muscle weakness and ultimately cardiac and respiratory failure. Immediate family members are often primary caregivers of individuals with a dystrophinopathy.
  
  Methods
  
  We explored the impact of this role by inviting primary caregivers (n = 209) of males diagnosed with childhood-onset dystrophinopathy who were identified by the Muscular Dystrophy Surveillance, Tracking, and Research Network (MD STARnet) to complete a mailed questionnaire measuring perceived social support and stress, spirituality, and family quality of life (FQoL). Bivariate and multivariate analyses examined associations between study variables using the Double ABCX model as an analytic framework.
  
  Results
  
  Higher stressor pile-up was associated with lower perceived social support (r = -0.29, p < .001), availability of supportive family (r = -0.30, p < .001) or non-family (r = -0.19, p < .01) relationships, and higher perceived stress (r = 0.33, p < .001); but not with spirituality (r = -0.14, p > 0.05). FQoL was positively associated with all support measures (correlations ranged from: 0.25 to 0.58, p-values 0.01–0.001) and negatively associated with perceived stress and control (r = -0.49, p < .001). The association between stressor pile-up and FQoL was completely mediated through global perceived social support, supportive family relationships, and perceived stress and control; supportive non-family relationships did not remain statistically significant after controlling for other mediators.
  
  Conclusions
  
  Findings suggest caregiver adaptation to a dystrophinopathy diagnosis can be optimized by increased perceived control, supporting family resources, and creation of a healthy family identity. Our findings will help identify areas for family intervention and guide clinicians in identifying resources that minimize stress and maximize family adaptation.
  
  
• Subjects:
  Adaptation, Psychological Adolescent Adult Becker Muscular Dystrophy Caregivers Child Child, Preschool Comorbidity Duchenne Muscular Dystrophy Dystrophinopathy Female Health Resources Humans Infant Internal-External Control Life Change Events Male Middle Aged Models, Psychological Muscular Dystrophies Muscular Dystrophy, Duchenne Quality Of Life Research Retrospective Studies Social Support Stress, Psychological Surveys And Questionnaires Young Adult

  More +
• Source:
  Health Qual Life Outcomes. 15.
• Pubmed ID:
  28187773
• Pubmed Central ID:
  PMC5303295
• Document Type:
  Journal Article
• Funding:
  U01 DD000187/DD/NCBDD CDC HHS/United States ; U01 DD000189/DD/NCBDD CDC HHS/United States ; U01 DD000191/DD/NCBDD CDC HHS/United States ; U01 DD000190/DD/NCBDD CDC HHS/United States ; U01 DD000831/DD/NCBDD CDC HHS/United States
• Volume:
  15
• Collection(s):
  CDC Public Access
• Main Document Checksum:
  urn:sha256:82bc615e5b2cddca4e55d8fa28015c1fc23d13a54eea4dff901661720ffc42c7
• Download URL:
  https://stacks.cdc.gov/view/cdc/44401/cdc_44401_DS1.pdf
• File Type:
  [PDF - 869.17 KB ]