Al-Bluwi Ghada S M, AlNababteh Asma H, Östlundh Linda, Al-Shamsi Saif, Al-Rifai Rami H
Department of Internal Medicine, College of Medicine and Health Sciences, United Arab Emirates University, Al-Ain, United Arab Emirates.
Institute of Public Health, College of Medicine and Health Sciences, United Arab Emirates University, Al-Ain, United Arab Emirates.
Front Med (Lausanne). 2021 Jun 17;8:674896. doi: 10.3389/fmed.2021.674896. eCollection 2021.
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. 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. 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, , 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. 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.
乳糜泻（CD）是一种多因素自身免疫性疾病，研究报告称特纳综合征（TS）患者有患CD的风险。本系统评价和荟萃分析旨在量化TS患者中CD的加权患病率，并确定TS与CD之间关联的加权强度。从四个电子数据库（PubMed、Scopus、科学网和Embase）中检索了1991年1月至2019年12月发表的研究。两名独立评审员按照特定的纳入标准和数据提取计划确定符合条件的研究并提取相关数据。使用随机效应模型确定合并的、总体和亚组CD患病率，并使用元回归研究异质性来源。在总共1116条筛选出的引文中，36项符合条件的研究纳入了定量综合分析。近三分之二的研究（61.1%）来自欧洲国家。在6291名接受CD检测的TS患者中，241人被诊断为CD，CD的粗患病率为3.8%。瑞典和德国分别报告了最高和最低的CD患病率，分别为20.0%和0.0%。估计总体加权CD患病率为4.5%（95%置信区间[CI]，3.3 - 5.9，I² = 67.4%）。TS患者中基于血清学的加权CD患病率（3.4%，95%CI = 1.
