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

Natalia Frishman, Kristin Caspers Conway, Jennifer Andrews, Jacob Oleson, Katherine Mathews, Emma Ciafaloni, Joyce Oleszek, Molly Lamb, Dennis Matthews, Pangaja Paramsothy, Lowell McKirgan, Paul Romitti

Research output: Contribution to journalArticlepeer-review

10 Scopus citations

Abstract

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.

Original languageEnglish (US)
Article number33
JournalHealth and Quality of Life Outcomes
Volume15
Issue number1
DOIs
StatePublished - Feb 10 2017
Externally publishedYes

Keywords

  • Becker muscular dystrophy
  • Caregivers
  • Duchenne muscular dystrophy
  • Dystrophinopathy
  • Muscular dystrophies
  • Quality of life

ASJC Scopus subject areas

  • Public Health, Environmental and Occupational Health

Fingerprint

Dive into the research topics of 'Perceived quality of life among caregivers of children with a childhood-onset dystrophinopathy: A double ABCX model of caregiver stressors and perceived resources'. Together they form a unique fingerprint.

Cite this