Abstract
RATIONALE: Ameloblastic carcinoma is defined as an ameloblastoma in which there is histological evidence of malignancy in primary tumour or recurrent tumour regardless if it has metastasised or not. It is aggressive in nature. PATIENT CONCERNS: The patient presented with a painful swelling associated with restricted mouth opening. DIAGNOSIS: This is the clinical profile of a patient who has presented with a recurrent ameloblastic carcinoma. TREATMENT: Histological features of the excised specimen resemble ameloblastic carcinoma constituting cytological atypia with tall columnar cells. OUTCOME: Excision of tumour was done. Reconstruction was done using pectoralis major myocutaneous flap. TAKE-AWAY LESSONS: Ameloblastic carcinoma is an aggressive tumour and constitutes destruction and distant metastatic spread. Hence, aggressive resection is the choice of treatment along with a long-term follow-up for better quality of life.