Stone, Julia
(2018)
Caregiver Perceptions and quality of life in adolescents with Duchenne Muscular Dystrophy.
Master's Thesis, University of Pittsburgh.
(Unpublished)
Abstract
Duchenne muscular dystrophy (DMD) is a rare neuromuscular disorder characterized by early proximal muscle weakness and progressive loss of muscle function that primarily affects males. Although there is currently no cure for the disease, there is ongoing research into treatments to slow the disease progression, which could impact overall quality of life (QoL) for boys and men with DMD. Many early studies on QoL in DMD were conducted using parent-proxy reports but more recent research has suggested that boys with DMD might perceive their QoL differently than their caregivers. The purpose of this study was to determine if parents of adolescents with DMD perceive their child’s QoL the same as the boys perceive their own QoL and to determine if specific aspects of DMD (glucocorticoid use, loss of ambulation, noninvasive respiratory support, and inability to self-feed) affect these perceptions. This study analyzed data from PedsQLTM 4.0 Generic Core Scale surveys and the PedsQLTM 3.0 Neuromuscular Module administered to participants ages 11-17 years old and their caregivers through the Cooperative International Neuromuscular Research Group (CINRG) Duchenne Natural History Study (DNHS). Overall, parents reported lower physical QoL scores (p<0.003) and higher emotional (p=0.049), social (p<0.001), and school-functioning (p<0.022) QoL scores compared to their sons. Older participants and their caregivers both reported higher physical QoL scores than younger participant/parent-proxy reports (p=0.017, p=0.035). Participants taking glucocorticoids reported lower physical QoL than participants not taking glucocorticoids (p<0.001). There was no difference in participant reported total QoL before and after both loss of ambulation and starting non-invasive respiratory support. The results suggest that adolescents with DMD have more positive perceptions regarding their physical QoL and more negative perceptions regarding their psychosocial QoL when compared to their parents, indicating a difference between adolescent experiences and parent perceptions. The results also suggest that the PedsQLTM 4.0 survey may not appropriately capture physical QoL in adolescents with DMD. Improving population health and QoL is an important public health goal, and the results of this study provides information that may help guide interventions to improve the QoL for adolescents with DMD.
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Details
| Item Type: |
University of Pittsburgh ETD
|
| Status: |
Unpublished |
| Creators/Authors: |
|
| ETD Committee: |
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| Date: |
28 June 2018 |
| Date Type: |
Publication |
| Defense Date: |
9 April 2018 |
| Approval Date: |
28 June 2018 |
| Submission Date: |
2 April 2018 |
| Access Restriction: |
No restriction; Release the ETD for access worldwide immediately. |
| Number of Pages: |
79 |
| Institution: |
University of Pittsburgh |
| Schools and Programs: |
School of Public Health > Genetic Counseling |
| Degree: |
MS - Master of Science |
| Thesis Type: |
Master's Thesis |
| Refereed: |
Yes |
| Uncontrolled Keywords: |
Duchenne Muscular Dystrophy, DMD, Quality of Life |
| Date Deposited: |
28 Jun 2018 20:15 |
| Last Modified: |
03 Jul 2018 13:39 |
| URI: |
http://d-scholarship.pitt.edu/id/eprint/34019 |
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