Background: Ameloblastoma, one of the most common odontogenic tumors, although classified as benign yet shows strong predilection for local infiltration. This leads to the recurrence of the tumor after surgical resection (more so when treated conservatively). Recurrent cases may progress toward malignant transformations on rare occasions, thus repeated attempts of conservative surgery in these should be avoided. Case Presentation: We present two cases of ameloblastoma that recurred after limited resection. One of them was showing focal area of cellular atypia denoting its path toward malignant transformation. Both the cases were managed with aggressive radical resection and suitable reconstruction. Conclusion: Tumor histology, anatomical location, and adequacy of tumor resection with safety margins are various factors which influence the recurrence of tumor and thus are to be considered along with the possibility of malignant transformation while formulating a treatment plan for revision cases. An individualized decision-making approach should be adopted for every case and long-term follow-up is necessary to avoid unacceptable morbidity due to extensive recurrences.

Keywords: Recurrent ameloblastoma, mandibular, maxillary, free fibular flap, malignant ameloblastoma, mandibular reconstruction.