Prioritizing Parental Worry Associated with Duchenne Muscular Dystrophy Using Best-Worst Scaling

Holly Landrum Peay, I. L. Hollin, J. F.P. Bridges

Research output: Contribution to journalArticle

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 languageEnglish (US)
Pages (from-to)305-313
Number of pages9
JournalJournal of Genetic Counseling
Volume25
Issue number2
DOIs
StatePublished - Apr 1 2016

Keywords

  • Best-worst scaling
  • Disease impact
  • Duchenne muscular dystrophy
  • Worry

ASJC Scopus subject areas

  • Genetics(clinical)

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