Adenomatoid Odontogenic Tumor (AOT) is a benign tumor of rare recurrence, most prevalent in females and anterior region of the maxilla. We report an atypical occurrence of follicular AOT in the anterior mandibular region in male patient and its surgical approach. Twenty-six years-old male presented swelling in the right anterior mandibular region. Radiography showed a radiolucent well-circumscribed lesion, associated with unerupted tooth right permanent mandibular canine. Incisional biopsy and installation of decompressive device were performed. First histopathological diagnosis was not conclusive suggesting dentigerous cyst, however follow-up of 1-year showed non-regression of the lesion. A new approach was performed through enucleation and curettage, and the second diagnosis was conclusive of AOT. At 18-months follow-up, bone formation was observed and patient presented no complaints. Surgical enucleation is the treatment indicated for AOT, which is easily removed due to its fibrous capsule. Due to its mimetic features, AOT can be misdiagnosed. Therefore, the correct diagnosis is important to offer the patient adequate treatment.
