Ameloblastoma is a slow growing odontogenic epithelial tumor of the jaw, however maxillary ameloblastoma is a rare histopathological entity. This tumor arises from odontogenic epithelium of emb ryona l to oth element and is most frequent in the mandibular molar region, with only 20% occurring in the maxilla. Of the various histological patterns of amelo blas- toma, the granular cell type is extremely rare, accounting for 4% of all reported cases of amelo blastomas. We report a rare case of multicystic g ranular cell ameloblastoma of maxilla in a 15 years old male patient who presented clinically as slow grow- ing painful swelling on left side of the face. Histological examina- tion of the excised tumor revealed islands comprising of cells with extensive granular cell transformation, surrounded by peri- pheral ameloblast-like cells. Immunohistochemistry showed positivity for pancytokeratin while negative staining was seen for S-100 and other neural and muscle markers, thereby confirming the diagnosis of granular cell ameloblastoma.
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