Ameloblastomas are locally aggressive jaw tumors with a high propensity for recurrence that are believed to arise from remnants of odontogenic epithelium, lining of odontogenic cysts and basal layer of overlying oral mucosa. They can occur in either the maxilla or mandible at nearly any age but most frequently are discovered as a painless expansion in the mandible of patients in their 20s-40s. Histopathologically, the follicular and plexiform patterns are the most common. When extensive squamous metaplasia, often associated with keratin formation occurs in central portions of the epithelial islands of follicular ameloblastoma, the term acanthomatous is sometimes applied. Here we present a case of acanthomatous ameloblastoma in a 20 year old patient. Key words:Ameloblastoma, Aacanthomatous, Mandibular Tumors. How to cite this article: Geeta Singh, Rashmi Agarwal, Vimlesh Kumar, Deepak Passi. Acanthomatous Ameloblastoma- A Case Report. J Int Oral Health 2013; 5(2):60-64..