Ameloblastoma is the most common aggressive benign odontogenic tumor of the 
jaws. Ameloblastoma is a benign epithelial odontogenic tumor that typically 
arises in the mandible or maxilla or, rarely, in the immediate adjacent soft 
tissues. A clinical, radiographic and histopathological report is presented of a 
case of acanthomatous ameloblastoma in relation to molar in the left mandible of 
a 30-year-old healthy male. The histopathological examination of the removed 
specimen revealed the histopathological pattern of an acanthomatous 
ameloblastoma. The radiographic appearance of the lesion showed the presence of 
multilocular radiolucencies, which were crossing the midline, which is rarely 
found in ameloblastoma. Due to its rarity and lack of data, we take this 
opportunity to present a world first case of acanthomatous ameloblastoma which 
was crossing the midline.