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

doi:10.1186/s12955-017-0612-1

Back

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

Journal article

Open access

Peer reviewed

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, …

Health and quality of life outcomes, Vol.15(1), pp.33-33

02/10/2017

DOI: 10.1186/s12955-017-0612-1

PMCID: PMC5303295

PMID: 28187773

Files and links (1)

url

https://doi.org/10.1186/s12955-017-0612-1View

Published (Version of record) Open Access

Abstract

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. 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. 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. 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.

Comorbidity

Caregivers - psychology

Humans

Life Change Events

Middle Aged

Social Support

Child, Preschool

Models, Psychological

Infant

Male

Muscular Dystrophy, Duchenne - psychology

Young Adult

Adolescent

Internal-External Control

Adult

Female

Surveys and Questionnaires

Quality of Life - psychology

Retrospective Studies

Muscular Dystrophy, Duchenne - therapy

Child

Health Resources

Stress, Psychological - complications

Adaptation, Psychological

Details

Title: Subtitle: Perceived quality of life among caregivers of children with a childhood-onset dystrophinopathy: a double ABCX model of caregiver stressors and perceived resources
Creators: Natalia Frishman - Present address: General Dynamics Information Technology, Coralville, IA, USA
Kristin Caspers Conway - Department of Epidemiology, The University of Iowa, Iowa City, USA
Jennifer Andrews - Department of Pediatrics, The University of Arizona, Tucson, USA
Jacob Oleson - Department of Biostatistics, The University of Iowa, Iowa City, USA
Katherine Mathews - Departments of Pediatrics and Neurology, The University of Iowa, Iowa City, USA
Emma Ciafaloni - Departments of Neurology and Pediatrics, University of Rochester Medical Center, Rochester, USA
Joyce Oleszek - Department of Physical Medicine and Rehabilitation, University of Colorado and Children's Hospital Colorado, Aurora, USA
Molly Lamb - Department of Epidemiology, Colorado School of Public Health, Aurora, USA
Dennis Matthews - Department of Physical Medicine and Rehabilitation, University of Colorado and Children's Hospital Colorado, Aurora, USA
Pangaja Paramsothy - National Center on Birth Defects and Developmental Disabilities, Centers for Disease Control and Prevention, Atlanta, USA
Lowell McKirgan - Department of Epidemiology, The University of Iowa, Iowa City, USA
Paul Romitti - Departments of Epidemiology and Biostatistics and Interdisciplinary Program in Toxicology, The University of Iowa, College of Public Health, S416 CPHB, 145 N Riverside Dr, Iowa City, IA, 52242, USA. paul-romitti@uiowa.edu
Resource Type: Journal article
Publication Details: Health and quality of life outcomes, Vol.15(1), pp.33-33
DOI: 10.1186/s12955-017-0612-1
PMID: 28187773
PMCID: PMC5303295
NLM abbreviation: Health Qual Life Outcomes
ISSN: 1477-7525
eISSN: 1477-7525
Publisher: England
Grant note: U01 DD000189 / NCBDD CDC HHS U01 DD001119 / NCBDD CDC HHS U01 DD000187 / NCBDD CDC HHS U01 DD000191 / NCBDD CDC HHS U01 DD000831 / NCBDD CDC HHS U01 DD000190 / NCBDD CDC HHS
Language: English
Date published: 02/10/2017
Academic Unit: Neurology; Stead Family Department of Pediatrics; Epidemiology; Iowa Neuroscience Institute; Biostatistics; Neurology (Pediatrics)
Record Identifier: 9983996186602771

Metrics

19 Record Views

27 Times Cited - Web of Science

See more details