Prevalence of developmental defects of enamel in individuals with celiac disease: A Systematic Review and Meta-analysis

ABSTRACT Objective To estimate the pooled prevalence of developmental defects of enamel (DDE) in individuals with celiac disease. Design A systematic search was conducted in PubMed/MEDLINE, Web of Science, Scopus, Embase, the Virtual Health Library, and grey literature. Observational studies reporting prevalence data on DDE in individuals with celiac disease were included. Risk of bias was assessed using the Joanna Briggs Institute Checklist for Prevalence Studies. Random-effects meta-analyses were performed to estimate pooled prevalence values, and subgroup analyses were performed to explore potential sources of heterogeneity. Results Of 45 included studies (n=3,045), 44 contributed to the meta-analysis (n=2,996). The overall pooled prevalence of DDE was 52.0% (95% CI: 45.0%–60.0%), with substantial heterogeneity (I² = 87.2%) and a wide 95% prediction interval (13.0%–89.0%). According to Aine’s classification, the pooled prevalence was 33.0% (95% CI: 29.0%–38.0%) for Grade I, 11.0% (95% CI: 9.0%–14.0%) for Grade II, 2.0% (95% CI: 1.0%–3.0%) for Grade III, and < 1% (95% CI: 0.0%–1.0%) for Grade IV. Subgroup analyses demonstrated a statistically significant difference in prevalence estimates based on the diagnostic criteria used for DDE assessment (p = 0.003) , whereas geographic location and medical diagnostic criteria for celiac disease showed no statistically significant differences in prevalence estimates ( p = 0.261 and p = 0.512 , respectively). Conclusions DDEs are frequently observed among individuals with celiac disease. Separate grade-specific meta-analyses yielded higher pooled estimates for milder DDE categories. However, the estimated prevalence varies considerably between populations and differs significantly according to the diagnostic criteria used to define DDE.