OBJECTIVES: To assess the cost-effectiveness of tests for diagnosing celiac disease (CD). The following tests were assessed: biopsy; endomysial antibody (EMA); antigliadin antibody (AGA); deaminated gliadin peptide (DGP); tissue transglutaminase (TTG); and panel of tests. METHODS: A decision analysis was constructed to compare costs and outcomes based on sensitivity, specificity and prevalence estimates from a primary systematic literature review. Study results were pooled using a bivariate, binomial, generalized linear mixed model (SAS 9.2). The target population was adults and children experiencing symptoms consistent with CD. Biopsy was assumed to have sensitivity and specificity of 1 since it is considered the gold standard. In serologic testing strategies the positive cases (true or false) were assumed to be confirmed with biopsy. A family physician consult was incurred with serologic testing and a gastroenterologist consult was incurred with biopsy. The outcome of the analysis was expected costs and false negatives (FN). Costs were reported in 2010 CAD$. All analyses were performed using TreeAge Pro Suite 2009. RESULTS: Four strategies made up the efficiency frontier; IgGTTG, IgATTG, EMA and biopsy.  All other strategies were either strictly or extendedly dominated. IgGTTG was the least costly and least effective strategy ($178.95, 0.1553 FNs). Biopsy was the most costly and most effective strategy ($396.60, 0 FNs). The cost per FN avoided moving from IgGTTG to the other strategies sequentially on the efficiency frontier were $293, $369, $1401 for EMA, IgATTG and biopsy respectively. One-way sensitivity analyses did not change the ranking of strategies. CONCLUSIONS: All testing strategies with biopsy were cheaper than biopsy alone however they also resulted in more FNs. If a decision maker’s willingness to pay to avoid a FN result is $1401 or greater, then biopsy alone is the most cost-effective strategy. Otherwise, either IgGTTG, IgATTG or EMA is the most cost-effective strategy.