Adenomatoid odontogenic tumor (AOT) is one of the intraoral odontogenic tumors. It occurs predominantly in anterior maxillary area of child and young adults in association with impacted teeth. There is predilection for occurrence of the tumor in female over male, by a ratio of 2:1. The general treatment method is conservative surgical excision, and recurrence rate is extremely low to 0.2%. The case reported is a extra-follicular type AOT. Pre-operative biopsy reporter as ameloblastic odontoma. In consideration of the patient is an adolescent female, we decided conservative surgical method with modified marsupialization to prevent the impact of facial appearance and oral function by radical surgery. After three-year follow -up, the lesion site is free of tumor. Bone regeneration of the surgical defect was evident. The displaced teeth have retuned to a normal axis.
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