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Serum prolactin levels (SPL) were estimated in patients with celiac disease (CD), diagnosed as per ESPGAN criteria, on unrestricted gluten containing diet (group 1), as well as those consuming a gluten-free diet (GFD) (group 2). Forty-one children with CD, with 20 cases in group 1 (mean age 5.67 ± 2.14, range 2.5–10.5 years) and 21 cases in group 2 (mean duration of follow-up 2 years, range 1–4 years), and 41 age- and sex-matched controls were studied. Hyperprolactinemia was defined as serum prolactin >18 ng/ml in males and >24 ng/ml in females. Upper gastrointestinal endoscopic biopsy was performed in both study groups for initial and follow-up evaluation. Hyperprolactinemia was detected in all the patients of group 1 and one patient of group 2 who had severe villous atrophy. The SPL in group 1 (mean 48.3 ± 17.4; range 20–90 ng/ml) and group 2 (mean 18.3 ± 6.9; range 10–39 ng/ml) was significantly higher compared with the controls (mean 9.3 ± 4.5; range 2.4–20 ng/ml; p < 0.001). Among the patients with CD,mean SPL in group 1 was significantly higher than in group 2 (p < 0.001). In group 1, there was a positive correlation between SPL and duration of symptoms (p = 0.006, r = 0.768) and age of diagnosis (p < 0.001, r = 0.842). A positive correlation also existed in group 2 between SPL and degree of villous atrophy (p < 0.001, r = 0.71) and lamina propria infiltrate (p < 0.001, r = 0.568). Our results suggest that SPL has a significant correlation with activity of CD. Therefore serum prolactin estimation may provide an additional marker of disease activity in CD and may be a more viable option economically.