Ranking Future Outcomes Most Important to Parents of Children with Bronchopulmonary Dysplasia

Abstract

Objective: To assess which potential future outcomes are most important to parents of children with bronchopulmonary dysplasia, a disease that affects future respiratory, medical, and developmental outcomes for children born preterm.

Study design: We recruited parents from 2 children's hospitals' neonatal follow-up clinics and elicited their importance rating for 20 different potential future outcomes associated with bronchopulmonary dysplasia. These outcomes were identified and selected through a literature review and discussions with panels of parents and clinician stakeholders, via a discrete choice experiment.

Results: One hundred and 5 parents participated. Overall, parents ranked "Will my child be more vulnerable to other problems because of having lung disease?" as the most important outcome, with other respiratory health related outcomes also highly ranked. Outcomes related to child development and effects on the family were among the lowest ranked. Individually, parents rated outcomes differently, resulting in a broad distribution of importance scores for many of the outcomes.

Conclusions: The overall rankings suggest that parents prioritize future outcomes related to physical health and safety. Notably, for guiding research, some top-rated outcomes are not traditionally measured in outcome studies. For guiding individual counseling, the broad distribution of importance scores for many outcomes highlights the extent to which parents differ in their prioritization of outcomes.

Keywords: ethics/bioethics; neonatology; pulmonology.

Figure 1—

(online only) How questions display to participants in the discrete choice experiment. This figure displays a sample question as participants would have seen it.

Figure 2—

Average Importance Scores Across Respondents. This figure contains the point estimates of the mean importance score for each outcome. The 95% CI around each point estimate represents our level of confidence that the interval captures the true point estimate.

Figure 3—

(online only) Importance Scores Stratified by Hospital, Child’s Age, and Diaper/Food Insecurity. This figure contains the point estimates of the mean importance score for each outcome by stratified group. The 95% CI around each point estimate represents our level of confidence that the interval captures the true point estimate. Panel A is stratified by institution. Panel B is stratified by age (under 1 year versus 1 year or older). Panel C is stratified by resource insecurity (food and diaper security versus food and/or diaper insecurity). CHOP= Children’s Hospital of Philadelphia; NW= Nationwide Children’s Hospital.

Figure 3—

(online only) Importance Scores Stratified by Hospital, Child’s Age, and Diaper/Food Insecurity. This figure contains the point estimates of the mean importance score for each outcome by stratified group. The 95% CI around each point estimate represents our level of confidence that the interval captures the true point estimate. Panel A is stratified by institution. Panel B is stratified by age (under 1 year versus 1 year or older). Panel C is stratified by resource insecurity (food and diaper security versus food and/or diaper insecurity). CHOP= Children’s Hospital of Philadelphia; NW= Nationwide Children’s Hospital.

Figure 4—

Individual-Level Variation Across Outcome Importance Scores. This graph presents the interquartile range of outcomes, which represents the spread of the middle half of the scores in each outcome. The line within each box represents the median, the left edge of the box represents the 25th percentile, and the right edge represents the 75th percentile of scores. The whiskers extending out of the box represents minimum and maximum scores, except for outliers (more than 1.5 interquartile range beyond the 25th and 75th quartiles), which are represented with point markers.