Dataset Information

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

ABSTRACT:

Introduction

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.

Methods and analysis

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.

Ethics and dissemination

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.

Prospero registration number

CRD42019131881, dated 3 September 2019.

SUBMITTER: Al-Bluwi GSM

PROVIDER: S-EPMC7342855 | biostudies-literature | 2020 Jul

REPOSITORIES: biostudies-literature

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Publications

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

Al-Bluwi Ghada S M GSM Alnababteh Asma H AH Al-Shamsi Saif S Al-Rifai Rami H RH

BMJ open 20200706 7

<h4>Introduction</h4>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 ...[more]

PMID: 32636286

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Project description:BackgroundAlthough muscle strength has been reported to be associated with metabolic syndrome (MetS), the association is still controversial. Therefore, the purpose of this meta-analysis was to identify the association between handgrip strength (HGS) and MetS.MethodsOriginal research studies involving HGS and MetS from database inception to 20 May 2022 were selected from PubMed, Web of Science, Embase, China National Knowledge Infrastructure, Wanfang databases, and Chinese Biomedical Document Service System. The odds ratios (ORs) with 95% confidence intervals (CIs) of MetS for HGS were calculated using a random-effects model. A dose-response analysis was performed. Subgroup analysis and meta-regression were also conducted.ResultsThirty effect sizes (reported in 19 articles) with a total of 43,396 participants were included in this meta-analysis. All studies were considered to be of moderate-to-good quality. An inverse association between HGS (low vs. high) with MetS was shown (OR: 2.59, 95% CI: 2.06-3.25). Subgroup analyses demonstrated the pooled ORs of relative HGS (HGS/weight), relative HGS (HGS/BMI), and absolute HGS were 2.97 (95% CI: 2.37-3.71), 2.47 (95% CI: 1.08-5.63), and 1.34 (95% CI: 1.06-1.68), respectively. Dose-response analysis revealed a significant linear dose-response relationship between relative HGS (HGS/weight) and MetS in observational studies (0.1 HGS/weight: OR, 0.68; 95% CI: 0.62-0.75). Univariate meta-regression analysis indicated that country status, measuring tools of HGS, components of MetS, and diagnosed criteria of MetS explained 16.7%, 26.2%, 30.1%, and 42.3% of the tau-squared in the meta-regression, respectively.ConclusionThe results of the current meta-analysis indicated that lower HGS is associated with a higher risk of MetS. A linear dose-response association between lower relative HGS (HGS/weight) and increased prevalence of MetS was found. Accordingly, a lower HGS is a significant predictor of MetS.Systematic review registration[https://www.crd.york.ac.uk/PROSPERO/], identifier [CRD42021276730].

Project description:Introduction: Celiac disease (CD) is a multifactorial autoimmune disorder, and studies have reported that patients with Turner syndrome (TS) are at risk for CD. This systematic review and meta-analysis aimed to quantify the weighted prevalence of CD among patients with TS and determine the weighted strength of association between TS and CD. Methods: Studies published between January 1991 and December 2019 were retrieved from four electronic databases: PubMed, Scopus, Web of Science, and Embase. Eligible studies were identified and relevant data were extracted by two independent reviewers following specific eligibility criteria and a data extraction plan. Using the random-effects model, the pooled, overall and subgroup CD prevalence rates were determined, and sources of heterogeneity were investigated using meta-regression. Results: Among a total of 1,116 screened citations, 36 eligible studies were included in the quantitative synthesis. Nearly two-thirds of the studies (61.1%) were from European countries. Of the 6,291 patients with TS who were tested for CD, 241 were diagnosed with CD, with a crude CD prevalence of 3.8%. The highest and lowest CD prevalence rates of 20.0 and 0.0% were reported in Sweden and Germany, respectively. The estimated overall weighted CD prevalence was 4.5% (95% confidence interval [CI], 3.3-5.9, I 2, 67.4%). The weighted serology-based CD prevalence in patients with TS (3.4%, 95% CI, 1.0-6.6) was similar to the weighted biopsy-based CD prevalence (4.8%; 95% CI, 3.4-6.5). The strength of association between TS and CD was estimated in only four studies (odds ratio 18.1, 95% CI, 1.82-180; odds ratio 4.34, 95% CI, 1.48-12.75; rate ratio 14, 95% CI, 1.48-12.75; rate ratio 42.5, 95% CI, 12.4-144.8). Given the lack of uniformity in the type of reported measures of association and study design, producing a weighted effect measure to evaluate the strength of association between TS and CD was unfeasible. Conclusion: Nearly 1 in every 22 patients with TS had CD. Regular screening for CD in patients with TS might facilitate early diagnosis and therapeutic management to prevent adverse effects of CD such as being underweight and osteoporosis.

Dataset Information

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

Introduction

Methods and analysis

Ethics and dissemination

Prospero registration number

Publications

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

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