A Novel Patient-Derived Conceptual Model of the Impact of Celiac Disease in Adults: Implications for Patient-Reported Outcome and Health-Related Quality-of-Life Instrument Development

• Published in: Value in health Vol. 20; no. 4; pp. 637 - 643
• Main Authors: Leffler, Daniel A., MD, Acaster, Sarah, PSc, MSc, Gallop, Katy, MSc, Dennis, Melinda, RD, MS, Kelly, Ciarán P., MD, Adelman, Daniel C., MD
• Format: Journal Article
• Language: English
• Published: United States Elsevier Inc 01.04.2017; Elsevier Science Ltd
• Subjects: Activities of daily living; Adaptation, Psychological; Adult; Adults; Aged; Aged, 80 and over; Anxiety; Celiac disease; Celiac Disease - diagnosis; Celiac Disease - diet therapy; Celiac Disease - physiopathology; Celiac Disease - psychology; Clinical outcomes; conceptual model; Conceptual models; Cost of Illness; Cross-Sectional Studies; Data processing; Diet; Diet, Gluten-Free; Emotional well being; Female; Functional impairment; Gluten; Health status; Humans; Internal Medicine; Interviews as Topic; Male; Medical conditions; Middle Aged; Patient communication; Patient Compliance; Patient Reported Outcome Measures; patient-reported outcome; Patients; Physical ability; Qualitative Research; Quality of Life; Saturation; Sleep; Social activities; Social anxiety; Symptoms; Time Factors; Treatment Outcome; Young Adult; celiac disease; patient-reported outcome; conceptual model; quality of life
• ISSN: 1098-3015; 1524-4733
• DOI: 10.1016/j.jval.2016.12.016

Abstract Background Celiac disease is a chronic inflammatory condition with wide ranging effects on individual’s lives caused by a combination of symptoms and the burden of adhering to a gluten-free diet (GFD). Objectives To further understand patients’ experience of celiac disease, the impact it has on health-related quality of life (HRQOL), and to develop a conceptual model describing this impact. Methods Adults with celiac disease on a GFD reporting symptoms within the previous 3 months were included; patients with refractory celiac disease and confounding medical conditions were excluded. A semistructured discussion guide was developed exploring celiac disease symptoms and impact on patients’ HRQOL. An experienced interviewer conducted in-depth interviews. The data set was coded and analyzed using thematic analysis to identify concepts, themes, and the inter-relationships between them. Data saturation was monitored and concepts identified formed the basis of the conceptual model. Results Twenty-one participants were recruited, and 32 distinct gluten-related symptoms were reported and data saturation was reached. Analysis identified several themes impacting patients’ HRQOL: fears and anxiety, day-to-day management of celiac disease, physical functioning, sleep, daily activities, social activities, emotional functioning, and relationships. The conceptual model highlights the main areas of impact and the relationships between concepts. Conclusions Both symptoms and maintaining a GFD have a substantial impact on patient functioning and HRQOL in adults with celiac disease. The conceptual model derived from these data may help to design future patient-reported outcomes as well as interventions to improve the quality of life in an individual with celiac disease.