Ameloblastoma is a true benign odontogenic epithelial tumor, primarily arising in the jaw, and ranks as the second most prevalent odontogenic neoplasm following odontoma. Known for its diverse clinical, radiographic, and histological manifestations, ameloblastoma encompasses a wide spectrum of presentations. Unicystic ameloblastomas (UAs), a less common and generally less aggressive variant, appear as cystic lesions that can mimic ordinary jaw cysts in their clinical and radiologic features. However, histological examination reveals a distinctive odontogenic epithelium lining the cyst cavities, with some cases exhibiting luminal and mural growth. This article presents a unique case involving a 40-year-old female patient who experienced swelling in the right posterior maxilla for four months. Initially presumed to be a routine ameloblastoma, subsequent histopathological analysis identified it as an intraluminal type of UA with rare plexiform changes. It is characterized by a cystic space lined with ameloblast-like cells in a plexiform arrangement, setting it apart from other UA subtypes. Imaging often reveals a unilocular cystic appearance, which may obscure differential diagnosis by closely resembling other odontogenic cysts. The variations within ameloblastoma have always sparked considerable discussion, and we aim to elucidate the reasons behind this specific transformation and its distinctive characteristics.
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