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The authors report an unusual case of coexistence of antrochoanal polyp (ACP) and fibrous dysplasia (FD) in the maxilla. To the best of our knowledge, this condition has never been reported in the literature. The complete removal of the ACP through endoscopic sinus surgery was attained and FD was ultimately diagnosed by histopathological examination. It is hypothesized that chronic inflammation or irritation due to FD has an effect on the occurrence of ACP.