Cells with their nucleus pushed to the periphery in a crescent-like fashion by intracytoplasmic vacuole(s) are referred to as signet ring cells when the vacuole(s) contain mucin and signet ring-like cells when they are empty or contain a material other than mucin. Signet ring cells are commonly associated with adenocarcinomas. These cells are uncommon in the endometrium and have been found to be associated with both malignant and benign processes. We report the first case of signet ring-like cells within endometrial glands with Arias-Stella reaction.