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An Aneurysmal bone cyst (ABC) is a rare benign lesion seen as a locally destructive, rapidly expansile, mostly affecting long bones and vertebrae. The association of ABC with Cemento-ossifying fibroma (COF) is extremely rare with only few cases reported so far in maxilla. We are herewith reporting one such case of hybrid lesion in a 15-year old boy, who came with a solitary swelling of right maxilla and showing partial obliteration of buccal vestibular sulcus and paraesthesia over right cheek region. Coronal Computed tomography (CT) of Para nasal sinus (PNS) view revealed large expansile lesion with thinning of cortex in right maxillary sinus involving hard palate and right alveolar process. Histological features were confirmative of the diagnosis of Aneurysmal Bone Cyst of Maxilla secondary to Cemento-ossifying fibroma. Treatment consisted of the enucleation of the lesion with surgical curettage through an intraoral approach under general anaesthesia.

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