Abstract
BACKGROUND: The prevalence of coeliac disease (CD) is around 1%, but diagnosis is challenged by varied presentation and non-specific symptoms and signs. This study aimed to identify diagnostic indicators that may help identify patients at a higher risk of CD in whom further testing is warranted.
METHODS: International guidance for systematic review methods were followed and the review was registered at PROSPERO (CRD42020170766). Six databases were searched until April 2021. Studies investigating diagnostic indicators, such as symptoms or risk conditions, in people with and without CD were eligible for inclusion. Risk of bias was assessed using the QUADAS-2 tool. Summary sensitivity, specificity, and positive predictive values were estimated for each diagnostic indicator by fitting bivariate random effects meta-analyses.
FINDINGS: 191 studies reporting on 26 diagnostic indicators were included in the meta-analyses. We found large variation in diagnostic accuracy estimates between studies and most studies were at high risk of bias. We found strong evidence that people with dermatitis herpetiformis, migraine, family history of CD, HLA DQ2/8 risk genotype, anaemia, type 1 diabetes, osteoporosis, or chronic liver disease are more likely than the general population to have CD. Symptoms, psoriasis, epilepsy, inflammatory bowel disease, systemic lupus erythematosus, fractures, type 2 diabetes, and multiple sclerosis showed poor diagnostic ability. A sensitivity analysis revealed a 3-fold higher risk of CD in first-degree relatives of CD patients.
CONCLUSIONS: Targeted testing of individuals with dermatitis herpetiformis, migraine, family history of CD, HLA DQ2/8 risk genotype, anaemia, type 1 diabetes, osteoporosis, or chronic liver disease could improve case-finding for CD, therefore expediting appropriate treatment and reducing adverse consequences. Migraine and chronic liver disease are not yet included as a risk factor in all CD guidelines, but it may be appropriate for these to be added. Future research should establish the diagnostic value of combining indicators.
| Original language | English |
|---|---|
| Article number | e0258501 |
| Number of pages | 20 |
| Journal | PLoS ONE |
| Volume | 16 |
| Issue number | 10 |
| DOIs | |
| Publication status | Published - 25 Oct 2021 |
Bibliographical note
Funding Information:The work is funded by a Health Technology Assessment Programme (NIHR129020). The researchers were hosted by the National Institute for Health Research (NIHR) Applied Research Collaboration West (NIHR ARC West). Our funder requires us to retain copyright of this manuscript because this work also needs to be written in a report which will be published by the funder (as NIHR HTA report). The funders had no role in study design, data collection and analysis, decision to publish, or preparation of the manuscript. We would like to thank Jo Stubbs for her feedback from a patient perspective at the study design stage.
Publisher Copyright:
© 2021 Elwenspoek et al. This is an open access article distributed under the terms of the Creative Commons Attribution License, which permits unrestricted use, distribution, and reproduction in any medium, provided the original author and source are credited.
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