Aim: Anemia is a common complication in patients with inflammatory diseases such as ankylosing spondylitis. Recent data suggest that hepcidin is a major mediator of anemia with a central role in iron homeostasis and metabolism. The aim of this study was to evaluate the serum levels of hepcidin and its prohormone, prohepcidin, in patients with ankylosing spondylitis in comparison with healthy controls. Material and Method: Forty patients with ankylosing spondylitis (13 with anemia and 27 without anemia), 20 healthy adults were prospectively enrolled. Complete blood count, erythrocyte sedimentation rate, serum levels of hepcidin, prohepcidin, iron, total iron binding capacity, ferritin, transferrin, and C-reactive protein were measured. Results: Serum levels of prohepcidin and hepcidin were significantly higher in patients with ankylosing spondylitis compared to healthy controls (p<0.005). Positive correlation was determined between the serum hepcidin and prohepcidin levels in patients with ankylosing spondylitis (r=0.725, p<0.001). Discussion: To the best of our knowledge, this is the first report of serum levels of prohepcidin and hepcidin in the patients with ankylosing spondylitis. Serum levels of prohepcidin and hepcidin are closely associated with disease activity in patients with ankylosing spondylitis and might play a role in the pathogenesis of anemia of chronic disease associated with ankylosing spondylitis.