Abstract
Duchenne muscular dystrophy (DMD) is a progressive, fatal pediatric disorder with significant burden on parents. Assessing disease impact can inform clinical interventions. Best-worst scaling (BWS) was used to elicit parental priorities among 16 short-term, DMD-related worries identified through community engagement. Respondents viewed 16 subsets of worries, identified using a balanced, incomplete block design, and identified the most and least worrying items. Priorities were assessed using best-worst scores (spanning +1 to −1) representing the relative number of times items were endorsed as most and least worrying. Independent-sample t-tests compared prioritization of parents with ambulatory and non-ambulatory children. Participants (n = 119) most prioritized worries about weakness progression (BW score = 0.64) and getting the right care over time (BW = 0.25). Compared to parents of non-ambulatory children, parents of ambulatory children more highly prioritized missing treatments (BW = 0.31 vs. 0.13, p <0.001) and being a good enough parent (BW = 0.06 vs. −0.08, p = 0.010), and less prioritized child feeling like a burden (BW = −0.24 vs. −0.07, p <0.001). Regardless of child’s disease stage, caregiver interventions should address the emotional impact of caring for a child with a progressive, fatal disease. We demonstrate an accessible, clinically-relevant approach to prioritize disease impact using BWS, which offers an alternative to the use of traditional rating/ranking scales.
| Original language | English (US) |
|---|---|
| Pages (from-to) | 305-313 |
| Number of pages | 9 |
| Journal | Journal of Genetic Counseling |
| Volume | 25 |
| Issue number | 2 |
| DOIs | |
| State | Published - Apr 1 2016 |
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Keywords
- Best-worst scaling
- Disease impact
- Duchenne muscular dystrophy
- Worry
ASJC Scopus subject areas
- Genetics(clinical)
Cite this
Prioritizing Parental Worry Associated with Duchenne Muscular Dystrophy Using Best-Worst Scaling. / Peay, Holly Landrum; Hollin, I. L.; Bridges, J. F P.
In: Journal of Genetic Counseling, Vol. 25, No. 2, 01.04.2016, p. 305-313.Research output: Contribution to journal › Article
}
TY - JOUR
T1 - Prioritizing Parental Worry Associated with Duchenne Muscular Dystrophy Using Best-Worst Scaling
AU - Peay, Holly Landrum
AU - Hollin, I. L.
AU - Bridges, J. F P
PY - 2016/4/1
Y1 - 2016/4/1
N2 - Duchenne muscular dystrophy (DMD) is a progressive, fatal pediatric disorder with significant burden on parents. Assessing disease impact can inform clinical interventions. Best-worst scaling (BWS) was used to elicit parental priorities among 16 short-term, DMD-related worries identified through community engagement. Respondents viewed 16 subsets of worries, identified using a balanced, incomplete block design, and identified the most and least worrying items. Priorities were assessed using best-worst scores (spanning +1 to −1) representing the relative number of times items were endorsed as most and least worrying. Independent-sample t-tests compared prioritization of parents with ambulatory and non-ambulatory children. Participants (n = 119) most prioritized worries about weakness progression (BW score = 0.64) and getting the right care over time (BW = 0.25). Compared to parents of non-ambulatory children, parents of ambulatory children more highly prioritized missing treatments (BW = 0.31 vs. 0.13, p <0.001) and being a good enough parent (BW = 0.06 vs. −0.08, p = 0.010), and less prioritized child feeling like a burden (BW = −0.24 vs. −0.07, p <0.001). Regardless of child’s disease stage, caregiver interventions should address the emotional impact of caring for a child with a progressive, fatal disease. We demonstrate an accessible, clinically-relevant approach to prioritize disease impact using BWS, which offers an alternative to the use of traditional rating/ranking scales.
AB - Duchenne muscular dystrophy (DMD) is a progressive, fatal pediatric disorder with significant burden on parents. Assessing disease impact can inform clinical interventions. Best-worst scaling (BWS) was used to elicit parental priorities among 16 short-term, DMD-related worries identified through community engagement. Respondents viewed 16 subsets of worries, identified using a balanced, incomplete block design, and identified the most and least worrying items. Priorities were assessed using best-worst scores (spanning +1 to −1) representing the relative number of times items were endorsed as most and least worrying. Independent-sample t-tests compared prioritization of parents with ambulatory and non-ambulatory children. Participants (n = 119) most prioritized worries about weakness progression (BW score = 0.64) and getting the right care over time (BW = 0.25). Compared to parents of non-ambulatory children, parents of ambulatory children more highly prioritized missing treatments (BW = 0.31 vs. 0.13, p <0.001) and being a good enough parent (BW = 0.06 vs. −0.08, p = 0.010), and less prioritized child feeling like a burden (BW = −0.24 vs. −0.07, p <0.001). Regardless of child’s disease stage, caregiver interventions should address the emotional impact of caring for a child with a progressive, fatal disease. We demonstrate an accessible, clinically-relevant approach to prioritize disease impact using BWS, which offers an alternative to the use of traditional rating/ranking scales.
KW - Best-worst scaling
KW - Disease impact
KW - Duchenne muscular dystrophy
KW - Worry
UR - http://www.scopus.com/inward/record.url?scp=84961199698&partnerID=8YFLogxK
UR - http://www.scopus.com/inward/citedby.url?scp=84961199698&partnerID=8YFLogxK
U2 - 10.1007/s10897-015-9872-2
DO - 10.1007/s10897-015-9872-2
M3 - Article
VL - 25
SP - 305
EP - 313
JO - Journal of Genetic Counseling
JF - Journal of Genetic Counseling
SN - 1059-7700
IS - 2
ER -