Papilliferous keratoameloblastoma is an extremely rare variant of ameloblastoma, a benign odontogenic tumor, with only seven cases reported in the English language literature. This variant presents with the metaplastic transformation of stellate reticulum-like cells to the extent of forming papillary structures exhibiting superficial keratinization of varying thickness. This paper describes the pathognomonic macroscopic features of this tumor observed during gross examination under the stereo zoom microscope that differentiate it from the other odontogenic tumors which have not been explored in the previously documented cases. Also, in this paper, a detailed comparison of the macroscopic features observed under the stereo zoom microscope during gross examination with the microscopic features of the histologic section has been described proving to be useful in the histological differential diagnosis of the keratinizing variants of ameloblastoma. © 2023 Elsevier B.V., All rights reserved.