The accuracy of diagnostic indicators for coeliac disease: a systematic review and meta-analysis
The accuracy of diagnostic indicators for coeliac disease: a systematic review and meta-analysis
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.
Elwenspoek, Martha M.C.
6fa131b1-b60f-4eb8-a6cb-f3dab87b845c
Jackson, Joni
1f1810c9-e2d2-4584-81ea-1f446c29db95
O’Donnell, Rachel
f23c0d06-dd99-4eb7-ace8-b3602fb20e30
Sinobas, Anthony
06f3f525-70b5-42c1-a670-15d020848e8a
Dawson, Sarah
1689f00c-c088-4e64-85f4-253e727b05ec
Everitt, Hazel
80b9452f-9632-45a8-b017-ceeeee6971ef
Gillett, Peter
5057d58f-fbdc-48e2-bcd7-f085418adbf3
Hay, Alastair D.
bfae9e44-ae9b-473c-923f-1dea50747023
Lane, Deborah L.
cfc4f3d8-344b-4273-849f-a8f4a7ae3884
Mallett, Susan
9f55a45b-6330-45e9-b817-2f69ebe32d5d
Robins, Gerry
e7370277-583f-48ab-a778-66dc8aabfd16
Watson, Jessica C.
50a1667e-fb12-4f60-ab18-50a631da215b
Jones, Hayley E.
22849f0d-6153-40f9-87a9-8a62f6d3b045
Whiting, Penny
13470a2b-00dd-4f25-88c6-7607e641c184
25 October 2021
Elwenspoek, Martha M.C.
6fa131b1-b60f-4eb8-a6cb-f3dab87b845c
Jackson, Joni
1f1810c9-e2d2-4584-81ea-1f446c29db95
O’Donnell, Rachel
f23c0d06-dd99-4eb7-ace8-b3602fb20e30
Sinobas, Anthony
06f3f525-70b5-42c1-a670-15d020848e8a
Dawson, Sarah
1689f00c-c088-4e64-85f4-253e727b05ec
Everitt, Hazel
80b9452f-9632-45a8-b017-ceeeee6971ef
Gillett, Peter
5057d58f-fbdc-48e2-bcd7-f085418adbf3
Hay, Alastair D.
bfae9e44-ae9b-473c-923f-1dea50747023
Lane, Deborah L.
cfc4f3d8-344b-4273-849f-a8f4a7ae3884
Mallett, Susan
9f55a45b-6330-45e9-b817-2f69ebe32d5d
Robins, Gerry
e7370277-583f-48ab-a778-66dc8aabfd16
Watson, Jessica C.
50a1667e-fb12-4f60-ab18-50a631da215b
Jones, Hayley E.
22849f0d-6153-40f9-87a9-8a62f6d3b045
Whiting, Penny
13470a2b-00dd-4f25-88c6-7607e641c184
Elwenspoek, Martha M.C., Jackson, Joni, O’Donnell, Rachel, Sinobas, Anthony, Dawson, Sarah, Everitt, Hazel, Gillett, Peter, Hay, Alastair D., Lane, Deborah L., Mallett, Susan, Robins, Gerry, Watson, Jessica C., Jones, Hayley E. and Whiting, Penny
(2021)
The accuracy of diagnostic indicators for coeliac disease: a systematic review and meta-analysis.
PLoS ONE, 16 (10 October), [e0258501].
(doi:10.1371/journal.pone.0258501).
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.
Text
PONE-D-21-25925_FTC accepted PLOS ONE Coeliac Paper Oct 2021
- Accepted Manuscript
Text
journal.pone.0258501
- Version of Record
More information
Accepted/In Press date: 28 September 2021
Published date: 25 October 2021
Additional Information:
Funding: 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.
Identifiers
Local EPrints ID: 453247
URI: http://eprints.soton.ac.uk/id/eprint/453247
ISSN: 1932-6203
PURE UUID: 376a6f53-3a36-4f6c-a590-5e82385de4c8
Catalogue record
Date deposited: 11 Jan 2022 17:46
Last modified: 17 Mar 2024 02:50
Export record
Altmetrics
Contributors
Author:
Martha M.C. Elwenspoek
Author:
Joni Jackson
Author:
Rachel O’Donnell
Author:
Anthony Sinobas
Author:
Sarah Dawson
Author:
Peter Gillett
Author:
Alastair D. Hay
Author:
Deborah L. Lane
Author:
Susan Mallett
Author:
Gerry Robins
Author:
Jessica C. Watson
Author:
Hayley E. Jones
Author:
Penny Whiting
Download statistics
Downloads from ePrints over the past year. Other digital versions may also be available to download e.g. from the publisher's website.
View more statistics
