Strength of the association between Turner syndrome and coeliac disease: protocol for a systematic review and meta-analysis

Abstract

<jats:sec><jats:title>Introduction</jats:title><jats:p>Coeliac disease (CD) is a genetic autoimmune disorder characterised by a permanent sensitivity to the gluten contained in some grains. Certain patient groups are considered high risk for the development of CD, including, but not limited to, those with chromosomal disorders such as Turner syndrome (TS). Here, we present a protocol for a systematic review and meta-analysis that aims to comprehensively summarise the literature, and quantitatively estimate the weighted strength of the association between TS and CD.</jats:p></jats:sec><jats:sec><jats:title>Methods and analysis</jats:title><jats:p>Our protocol follows the Preferred Reporting Items for Systematic Review and Meta-Analysis Protocols 2015 guidelines. We will search PubMed, Scopus, Web of Science and Embase databases for relevant articles. Variant and broad search terms will be selected for identifying epidemiological studies reporting on the crude and/or adjusted association between TS and CD. Retrieved citations will be screened, and data from the eligible research reports against specific eligibility criteria will be extracted. We will then assess the risk of bias associated with the eligible studies using the Newcastle-Ottawa Scale. The overall weighted strength of the pooled association will be quantified using the random-effects model.</jats:p></jats:sec><jats:sec><jats:title>Ethics and dissemination</jats:title><jats:p>This review will use data from published literature; hence, ethical approval will not be needed. The resulting review will be the first to produce a comprehensive synthesis of the strength of the association between TS and CD. The results will be disseminated through a peer-reviewed journal as well as in local and international conferences and symposiums. Results dissemination would help healthcare providers and policy-makers to make informed decisions regarding the diagnosis and management of CD in high-risk individuals.</jats:p></jats:sec><jats:sec><jats:title>PROSPERO registration number</jats:title><jats:p>CRD42019131881, dated 3 September 2019.</jats:p></jats:sec>

Prevalence of Celiac Disease in Patients With Turner Syndrome: Systematic Review and Meta-Analysis

“…This systematic review was conducted and reported following the 2009 Preferred Reporting Items for Systematic Review and Meta-Analysis guidelines (35) (Supplementary Table 1). The review followed a previously published protocol (36) that was also registered in PROSPERO (registration number, CRD42019131881). The published protocol was designed to estimate the strength of association between TS and CD.…”

“…A comprehensive strategy was designed to search four electronic databases: PubMed, Scopus, Web of Science, and Embase. The search string was developed by an expert librarian (LÖ) and is available in the published protocol (36) and in Supplementary Box 1, which contains the results and search details for all databases. The literature search was performed in December 2019 with no restrictions on language or region.…”

“…To be deemed eligible, an observational study had to provide quantitative or quantifiable information on the prevalence of CD and/or effect measure on the association between TS and CD regardless of the age of patients with TS screened for CD. Further information on the inclusion and exclusion criteria is available in the published protocol (36).…”

“…Relevant data were extracted from the studies that were deemed eligible. Data extraction was independently performed by two reviewers (GSM-AB and AH-N) following predefined data extraction parameters described in the published protocol (36), with minor amendments to extract data related to prevalence estimates. Discrepancies between the reviewers were discussed with a third reviewer (RH-A) and resolved by consensus.…”