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

Shayna Coburn; Paige J Trojanowski; Jack Vagadori; Pamela Hinds; Monique Germone; Edwin Liu; Randi Streisand; James Bost

doi:10.14309/ajg.0000000000003132

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

Am J Gastroenterol. 2024 Oct 14. doi: 10.14309/ajg.0000000000003132. Online ahead of print.

Authors

Shayna Coburn^{1

2}, Paige J Trojanowski¹, Jack Vagadori¹, Pamela Hinds^{1

2

3}, Monique Germone^{4

5}, Edwin Liu^{4

5}, Randi Streisand^{1

2}, James Bost^{1

2}

Affiliations

¹ Center for Translational Research, Children's National Hospital, Washington, District of Columbia, USA.
² The George Washington School of Medicine and Health Sciences, Washington, District of Columbia, USA.
³ Department of Nursing Science, Professional Practice & Quality, Children's National Hospital, Washington, District of Columbia, USA.
⁴ Digestive Health Institute, Children's Hospital Colorado, Aurora, Colorado, USA.
⁵ University of Colorado School of Medicine, Aurora, Colorado, USA .

PMID: 39401060
DOI: 10.14309/ajg.0000000000003132

Abstract

Introduction: 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.

Methods: 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).

Results: 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.

Discussion: 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.

Abstract

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