Background: Serous carcinoma arising in adenomyosis and adenomyotic cyst is very rare. Only 3 serous carcinomas\nand 5 serous endometrial intraepithelial carcinomas (EIC) have been reported to date.\nMethods: We reviewed the clinicopathological features of 2 serous carcinoma in uterine adenomyosis and 1 serous\nEIC in adenomyotic cyst of the cervical stump.\nResults: Case 1 had an endometrial serous carcinoma in the uterine myometrium and the left ovary. A minor\ncomponent of benign endometrial glands with minimal endometrial stroma was found in the uterine mass\nand the surrounding myometrium. Case 2 showed 3 small foci of serous carcinoma, serous EIC and endometrial\nglandular dysplasia (EmGD) in the adenomyosis. Scanty serous carcinoma was present in the endometrium without\nevidence of myometrial invasion. The eutopic endometrium in both case 1 and 2 had no evidence of neoplastic\nchanges after complete examination. Case 3 had 3 microscopic serous EICs in the adenomyotic cysts of the\ncervical stump. One EIC lesion coexisted with EmGD. No cancer was found in the endocervical tube although\nthe preoperative endocervical biopsy showed a poorly differentiated endometrioid carcinoma. Immunohistochemistry\ndemonstrated that serous carcinoma in case 1 and EIC in all 3 cases showed a characteristic pattern of p53 and p16\nover expression, high Ki67 index, and lack of WT1, ER and PR staining. EmGD in case 1 and 3 had a similar staining\npattern except a lower Ki67 index and the presence of ER expression.\nConclusions: We believe that this case series may expand our recognition on serous carcinoma arising in uterine\nadenomyosis/adenomyotic cyst including extra-uterine spread and the potential synchronous growth of carcinomas in\neutopic endometrium.
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