Strong associations between the polymorphisms of anthrax toxin receptor 2 (ANTXR2) and interleukin-1 receptor 2 (IL-1R2) and ankylosing spondylitis (AS) were identified through genome-wide scan by Australo-Anglo-American Spondyloarthritis Consortium very recently. However, the data are restricted to Caucasian patients in only the above report, and no replication study in other ethnic groups is published. Our study addresses this relationship in Chinese Han population. A cohort of 200 AS patients who satisfied the modified New York criteria and 200 ethnically matched healthy controls were recruited. All subjects were genotyped for rs4333130 of ANTXR2 and rs2310173 of IL-1R2. Genotyping was performed using the Sequenom MassARRAY system based on the chip-based matrix-assisted laser desorption ionization time-of-flight mass spectrometry platform. No association was observed between AS and ANTXR2 or IL-1R2 in present study, indicating that the polymorphisms in the ANTXR2 and IL-1R2 gene may not be regarded as major contributors to AS susceptibility in Chinese Han population. We also assumed that IL-1β might not be the key factor in the immunogenesis of AS.