Abstract
BackgroundSerous carcinoma arising in adenomyosis and adenomyotic cyst is very rare. Only 3 serous carcinomas and 5 serous endometrial intraepithelial carcinomas (EIC) have been reported to date.MethodsWe reviewed the clinicopathological features of 2 serous carcinoma in uterine adenomyosis and 1 serous EIC in adenomyotic cyst of the cervical stump.ResultsCase 1 had an endometrial serous carcinoma in the uterine myometrium and the left ovary. A minor component of benign endometrial glands with minimal endometrial stroma was found in the uterine mass and the surrounding myometrium. Case 2 showed 3 small foci of serous carcinoma, serous EIC and endometrial glandular dysplasia (EmGD) in the adenomyosis. Scanty serous carcinoma was present in the endometrium without evidence of myometrial invasion. The eutopic endometrium in both case 1 and 2 had no evidence of neoplastic changes after complete examination. Case 3 had 3 microscopic serous EICs in the adenomyotic cysts of the cervical stump. One EIC lesion coexisted with EmGD. No cancer was found in the endocervical tube although the preoperative endocervical biopsy showed a poorly differentiated endometrioid carcinoma. Immunohistochemistry demonstrated that serous carcinoma in case 1 and EIC in all 3 cases showed a characteristic pattern of p53 and p16 over expression, high Ki67 index, and lack of WT1, ER and PR staining. EmGD in case 1 and 3 had a similar staining pattern except a lower Ki67 index and the presence of ER expression.ConclusionsWe believe that this case series may expand our recognition on serous carcinoma arising in uterine adenomyosis/adenomyotic cyst including extra-uterine spread and the potential synchronous growth of carcinomas in eutopic endometrium.
Highlights
Serous carcinoma arising in adenomyosis and adenomyotic cyst is very rare
We present 2 serous carcinomas in uterine adenomyosis and 1 serous endometrial intraepithelial carcinomas (EIC) in adenomyotic cyst of the cervical stump
Sampson or Colman’s criteria for the diagnosis of carcinoma arising from adenomyosis were proposed early in 1950s [11, 12]. These includes: “1) the carcinoma must not be present in the normally situated endometrium or elsewhere in the pelvis; b) the carcinoma must be seen to arise from the epithelium within the adenomyosis rather than invasion from another source; and c) endometrial stromal cells must be seen to support a diagnosis of adenomyosis”
Summary
Serous carcinoma arising in adenomyosis and adenomyotic cyst is very rare. 3 serous carcinomas and 5 serous endometrial intraepithelial carcinomas (EIC) have been reported to date. Carcinomatous changes from the ectopic endometrial glands in endometriosis have been reported in many studies [1,2,3,4]. The predominant histotypes of these cases are endometrioid carcinoma and clear cell carcinoma [4,5,6]. Serous carcinoma arising in uterine adenomyosis is extremely rare. 3 reports containing 3 serous carcinomas and 5 serous endometrial intraepithelial carcinomas (EIC) have been reported to date [7,8,9,10]. We present 2 serous carcinomas in uterine adenomyosis and 1 serous EIC in adenomyotic cyst of the cervical stump
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