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Ameloblastoma is the second most common odontogenic neoplasm and is locally aggressive. Various treatment approaches exist, with resection involving safe margins associated with the lowest recurrence rates. However, ameloblastoma still has a propensity for delayed recurrences, necessitating lifelong follow-up for patients. Recurrences usually develop at the primary site of the tumor but have also been reported in reconstructive bone grafts. We present a unique case of a very unusual, delayed recurrence of mandibular ameloblastoma in the soft tissues of the neck, 16 years after the primary surgery. The recurrence presented as a large cystic swelling in the neck with a solid component, posing a diagnostic challenge for the team. The final histopathological examination after excision under general anesthesia confirmed it to be ameloblastoma.

Ameloblastoma is the second most common odontogenic neoplasm and is locally aggressive. Various treatment approaches exist, with resection involving safe margins associated with the lowest recurrence rates. However, ameloblastoma still has a propensity for delayed recurrences, necessitating lifelong follow-up for patients. Recurrences usually develop at the primary site of the tumor but have also been reported in reconstructive bone grafts. We present a unique case of a very unusual, delayed recurrence of mandibular ameloblastoma in the soft tissues of the neck, 16 years after the primary surgery. The recurrence presented as a large cystic swelling in the neck with a solid component, posing a diagnostic challenge for the team. The final histopathological examination after excision under general anesthesia confirmed it to be ameloblastoma.