Clear cell adenocarcinoma arising from adenomyosis mimicking leiomyoma: A case report

Abstract

The incidence of cancer from adenomyosis is rare. Previously, only two cases of clear cell adenocarcinoma (CCA) arising from adenomyosis have been reported in English literature. Here, we report a case of CCA arising from adenomyosis. A 52-year-old postmenopausal Korean woman presented with complaints of vaginal bleeding and back pain. Endometrial biopsy revealed endometrial polyp with atrophic change and transvaginal ultrasonography showed myomas with cystic change. Magnetic resonance imaging revealed a cystic degenerative mass consistent with leiomyoma in the posterior portion of uterus body. And the serum level of CA-125 was 17.7 U/mL. Hysterectomy revealed a yellow-ten solid mass in the myometrium that was diagnosed as CCA arising from adenomyosis. The tumor was mainly located in the myometrium and transition between adenomyosis and CCA along with endometrial stromal cell was identifi ed. Malignant tumor arising from adenomyosis could be considered as a differential diagnosis when the patient with adenomyosis and intact endometrial surface complained of vaginal bleeding.

Clear cell adenocarcinoma arising from adenomyotic cyst: A case report and literature review

“…12 Kumar and Anderson pointed out the necessity of a transition between the benign adenomyotic endometrial and carcinomatous glands to prove a diagnosis of ectopic endometriumderived adenocarcinoma. 11 In our case, although there were foci of isolated clear cell adenocarcinoma in the endometrium, the diagnosis was compatible with clear cell adenocarcinoma arising from adenomyosis because the tumor was mainly located in the adenomyotic cyst wall in the myometrium as in situ carcinoma with an obvious transition from nonneoplastic epithelium along with endometrial stromal cells. Furthermore, immunohistochemical analysis demonstrated that some of the epithelial cells of the adenomyotic cyst were ER À and p53 + , the same staining pattern as that of clear cell adenocarcinoma, suggesting that the epithelial cells of the adenomyotic cyst, which histologically appeared somewhat atypical but non-neoplastic, harbored early genetic abnormalities for carcinogenesis and precancerous cells, from which clear cell adenocarcinoma arose.…”

“…A PubMed search performed using the key words 'uterine clear cell adenocarcinoma' and 'adenomyosis' revealed five cases published between 1994 and 2011. [7][8][9][10][11] Herein, we report the sixth case of clear cell adenocarcinoma arising from adenomyosis (Table 1).…”

“…Postoperative histopathological findings fulfilled Sampson's or Colman's criteria for adenocarcinomaadenomyosis association: the carcinoma was not located in the endometrium or elsewhere in the pelvis; the carcinoma arose from the epithelium of the adenomyosis and did not invade from another source; and endometrial (adenomyotic) stromal cells were present to support a diagnosis of adenomyosis. 11 Other authors found a number of histologic features useful in identifying adenocarcinoma arising from adenomyosis, such as the presence of a smooth, round contour of surrounding myometrium, adenomyotic glands and endometrial-type stroma within the carcinoma foci and the absence of desmoplasia or an inflammatory response. 12 Kumar and Anderson pointed out the necessity of a transition between the benign adenomyotic endometrial and carcinomatous glands to prove a diagnosis of ectopic endometriumderived adenocarcinoma.…”

Endometrial Cancer Arising in Adenomyosis (EC-AIA): A Systematic Review

“…For the "unbiased study endpoints" domain, the risk of bias was low in 14 studies [13,14,17,18,20,24,[29][30][31][32][33]37,39,40,42], unclear in 11 studies [15,16,19,22,23,[26][27][28]34,35], and high in six studies [12,21,25,36,38,41]; in particular, in the studies at high risk of bias, Colman and Rosenthal's criteria were only partially fulfilled.…”

“…For the "patient selection" domain (when applicable), two studies were considered at low risk of bias [12,42], while two studies were judged as having unclear risk of bias [34,35]. For the "study endpoints" domain, the risk of bias was considered low in 15 studies [12][13][14]17,18,20,21,24,[29][30][31][32][33]37,41,42], while it was unclear in 16 studies [15,16,19,22,23,[25][26][27][28][34][35][36][38][39][40].…”