Value Health. 2024 Mar;27(3):301-312. doi: 10.1016/j.jval.2023.12.010. Epub 2023 Dec 26.

Edna Keeney ¹, Martha M C Elwenspoek ², Joni Jackson ³, Cristina Roadevin ⁴, Hayley E Jones ⁴, Rachel O'Donnell ², Athena L Sheppard ⁵, Sarah Dawson ⁴, Deborah Lane ⁶, Jo Stubbs ⁶, Hazel Everitt ⁷, Jessica C Watson ⁴, Alastair D Hay ⁴, Peter Gillett ⁸, Gerry Robins ⁹, Sue Mallett ¹⁰, Penny F Whiting ⁴, Howard Thom ⁴

Author information

¹Population Health Sciences, Bristol Medical School, University of Bristol, Bristol, England, UK. Electronic address: edna.keeney@cliftoninsight.co.uk.

²Population Health Sciences, Bristol Medical School, University of Bristol, Bristol, England, UK; The National Institute for Health Research Applied Research Collaboration West (NIHR ARC West), University Hospitals Bristol NHS Foundation Trust, Bristol, England, UK.

³The National Institute for Health Research Applied Research Collaboration West (NIHR ARC West), University Hospitals Bristol NHS Foundation Trust, Bristol, England, UK.

⁴Population Health Sciences, Bristol Medical School, University of Bristol, Bristol, England, UK.

⁵Population Health Sciences, Bristol Medical School, University of Bristol, Bristol, England, UK; The National Institute for Health Research Applied Research Collaboration West (NIHR ARC West), University Hospitals Bristol NHS Foundation Trust, Bristol, England, UK; Swansea University Medical School, Swansea University, Swansea, England, UK.

⁶Patient representative.

⁷Primary Care Research Centre, Population Sciences and Medical Education, University of Southampton, Southampton, England, UK.

⁸Paediatric Gastroenterology, Hepatology and Nutrition Department, Royal Hospital for Sick Children, Edinburgh EH9 1LF Scotland, England, UK.

⁹Department of Gastroenterology, York Teaching Hospital NHS Foundation Trust, York, England, UK.

¹⁰Centre for Medical Imaging, University College London, London, England, UK.

Abstract

Objectives: Celiac disease (CD) is thought to affect around 1% of people in the United Kingdom, but only approximately 30% are diagnosed. The aim of this work was to assess the cost-effectiveness of strategies for identifying adults and children with CD in terms of who to test and which tests to use.

Methods: A decision tree and Markov model were used to describe testing strategies and model long-term consequences of CD. The analysis compared a selection of pre-test probabilities of CD above which patients should be screened, as well as the use of different serological tests, with or without genetic testing. Value of information analysis was used to prioritize parameters for future research.

Results: Using serological testing alone in adults, immunoglobulin A (IgA) tissue transglutaminase (tTG) at a 1% pre-test probability (equivalent to population screening) was most cost-effective. If combining serological testing with genetic testing, human leukocyte antigen combined with IgA tTG at a 5% pre-test probability was most cost-effective. In children, the most cost-effective strategy was a 10% pre-test probability with human leukocyte antigen plus IgA tTG. Value of information analysis highlighted the probability of late diagnosis of CD and the accuracy of serological tests as important parameters. The analysis also suggested prioritizing research in adult women over adult men or children.

Conclusions: For adults, these cost-effectiveness results suggest UK National Screening Committee Criteria for population-based screening for CD should be explored. Substantial uncertainty in the results indicate a high value in conducting further research.