Development and Validation of a Pediatric Celiac Disease-Specific Quality of Life Measure.

Abstract

Quality of life (QOL) is critical in screening and management of chronic medical conditions, including celiac disease (CD). The aim of this project was to develop a CD-specific pediatric QOL measure (Celiac Disease Life Inventory of Family Experiences [CDLIFE]) with parallel self-report and parent-report forms by generating items through concept elicitation interviews, iterative refinement using cognitive debriefing interviews, and evaluating its psychometric properties and validity. Concept elicitation interviews were conducted to develop items (9 youth ages 8-19 years with CD; 10 parents of youth with CD), followed by cognitive interviews with additional stakeholders (3 youth with CD, 3 parents, and 8 clinicians) and item administration (parent/youth reports: n = 103/102). Analyses included response frequencies, internal consistency reliability, exploratory factor analyses, and correlations with related measures (Patient-Reported Outcomes Measurement Information System, Pediatric Quality of Life Inventory, and Gluten-Free Diet-Visual Analog Scale). From concept elicitation interviews, 42 youth and 45 parent items were developed. Cognitive debriefing interviews yielded 36 refined items. Psychometric analyses identified 15 items to remove due to ceiling/floor effects, poor item-to-test correlations, and youth-parent mismatch or conceptual mismatch. Total score internal consistency was high (αs = 0.89-0.90). A 4-factor model solution had the best fit (Social Impact, External Support, Adaptive Vigilance, Eating Behaviors/Adjustment) with a fifth single-item domain (Financial Resources). The final CDLIFE (21 items) total scores correlated with most related measures in expected directions for parent and youth forms. The CDLIFE may facilitate insight into CD-specific QOL for youth ages 2-18 years, capturing important dimensions of physical and socioemotional health. Administering the CDLIFE will help identify and track families needing support.