Arch Gynecol Obstet (2015) 291:399–402 DOI 10.1007/s00404-014-3413-3

GYNECOLOGIC ONCOLOGY

Decidualization of uterine adenomyoma in a pregnant woman: a case report with immunohistochemical study Tadashi Terada

Received: 20 January 2014 / Accepted: 7 August 2014 / Published online: 3 September 2014 Ó Springer-Verlag Berlin Heidelberg 2014

Abstract Background Decidualization of uterine adenomyoma has not been reported, to the best of the author’s knowledge. Aim To report a case of uterine adenomyoma with decidualization. Case report A 43-year-old pregnant woman with ‘‘myoma’’ underwent cesarean operation and ‘‘myomectomy’’ at 37 gestation weeks. The operation was successful, and the baby and mother were healthy. Grossly, the ‘‘myoma’’ measured 12 9 10 9 10 cm, and the consistency was firm. Microscopically, the tumor was adenomyoma consisting of smooth muscle bundles and endometrial islands. Characteristically, the endometrial stroma showed marked decidualization. An immunohistochemical study showed that the decidual cells were positive for pancytokeratin AE1/3, pancytokeratin CAM5.2, cytokeratin (CK)7, CK18, vimentin, CA125, CD10, estrogen receptor (ER), progesterone receptor (PgR), and Ki-67 (labeling 1 %). They were negative for CK34bE12, CK5/6, CK8, CK14, CK19, CK20, EMA, p63, desmin, a-smooth muscle actin, S100 protein, CK34, CD68, and p53. These results show that marked decidualization occurs in adenomyoma during pregnancy, and that the decidual cells are positive for pancytokeratin AE1/3, pancytokeratin CAM5.2, CK7, CK18, vimentin, CA125, CD10, ER, and PgR. Conclusion A rare case of uterine adenomyoma with decidualization is reported. Keywords Uterus  Pregnancy  Adenomyoma  Decidualization  Immunohistopathology T. Terada (&) Department of Pathology, Shizuoka City Shimizu Hospital, Miyakami 1231, Shimizu-Ku, Shizuoka 424-8636, Japan e-mail: [email protected]

Introduction The endometrium consists of endometrial glands and stroma. The stromal cells were compact and dense in the proliferative phase, and edematous and predeciduous in the secretory phase. During pregnancy, the stromal cells become epithelioid, plump and acidophilic decidual cells under the influence of estrogen and progesterone [1]. This decidualization occurs in ectopic endometrium (endometriosis) [2, 3]. Ectopic deciduosis has been also reported [4, 5]. Adenomyoma of the uterus is a tumor composed of smooth muscle cells and islands of endometrial gland and stroma [6]. However, to the best of the author’s knowledge, it is unknown whether decidualization occurs in the adenomyoma. Herein reported is a case of decidualization of adenomyoma in a pregnant woman. Extensive immunohistochemical study was also performed.

Case report A 43-year-old woman (para 2, gravida 2) with ‘‘myoma’’ became pregnant. The pregnancy course was good. At 37 weeks of gestation, she was treated by cesarean operation and ‘‘myomectomy’’, because she had ‘‘myoma’’. The operation was successful, and the baby and mother were healthy. Grossly, the ‘‘myoma’’ measured 12 9 10 9 10 cm, and the consistency was firm (Fig. 1). Microscopically, the tumor was adenomyoma consisting of smooth muscle bundles and endometrial islands (Fig. 2a). Characteristically, the endometrial stroma showed marked decidualization consisting of reddish, plump, and epithelioid cells (Fig. 2b). The endometrial glands were also seen in a small number, and lacked Arias-Stella phenomenon.

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Arch Gynecol Obstet (2015) 291:399–402 Table 1 Immunohistochemical findings of the uterine adenomyoma Decidua

Fig. 1 Gross features of the resected adenomyoma of the uterus. It is reddish, measured 12 9 10 9 10 cm, and firm in consistency

Muscle

Gland

CK AE1/3

???



???

CK CAM5.2

?



?

CK34BE12







CK5/6







CK7

?



?

CK8





?

CK14







CK18

??



??

CK19





??

CK20







EMA P63

– –

– –

? –

Vimentin

???

?



Desmin



?



HCG







ASMA



???



S100







CD34







CA125

?



?? –

CD68





CD10

???

?



ER

???

?

???

PgR

??

?

??

P53







Ki-67 (%)

1

1

2

CK cytokeratin, EMA epithelial membrane antigen, HCG human chorionic gonadotropin, ASMA a-smooth muscle antigen, ER estrogen receptor, PgR progesterone receptor

Fig. 2 Microscopic findings. a The tumor consisted of smooth muscles and islands of endometrial tissue with marked decidualization. HE, 950. b The decidual cells show plump, acidophilic and epithelioid features. Smooth muscle cells and endometrial glands are also seen. HE, 9200

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An immunohistochemical analysis was performed with the use of Dako’s envision method (Dako Corp, Glustrup, Denmark), as previously reported [7–20]. The immunohistochemical results are shown in Table 1. Immunohistochemically, the decidual cells were positive for pancytokeratin AE1/3 (Fig. 3a), pancytokeratin CAM5.2, cytokeratin (CK)7, CK18 (Fig. 3b), vimentin (Fig. 3c), CA125, CD10 (Fig. 3d), estrogen receptor (ER) (Fig. 3e), progesterone receptor (PgR), and Ki-67 (labeling 1 %). They were negative for CK34bE12, CK5/6, CK8, CK14, CK19, CK20, EMA, p63, desmin, a-smooth muscle actin, S100 protein, CK34, CD68, and p53. The glandular cells were positive for pancytokeratin AE1/3, pancytokeratin CAM5.2, CK7, CK8, CK18, CK19, EMA,ER, PgR, and Ki-67 (labeling 2 %). They were negative for CK34bE12, CK5/6, CK14, CK20, p63, vimentin, desmin, a-smooth muscle actin, S100 protein, CK34, CD68, CD10, and p53. The smooth muscle cells were positive for vimentin, desmin, a-smooth muscle actin, CD10, ER, PgR, and Ki-67

Arch Gynecol Obstet (2015) 291:399–402

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Fig. 3 Immunohistochemical findings of the decidual cells in uterine adenomyoma. The decidual cells are positive for cytokeratin AE1/3 (a), cytokeratin 10 (b), vimentin (c), CD10 (d), and estrogen receptor (e). a-e, 9200

(labeling = 1 %). They were negative for various CKs, EMA, p63, S100 protein, CD34, CA125, CD68, and p53. A pathological diagnosis of uterine adenomyoma with marked decidualization was made.

Discussion Decidualization of the endometrial tissue in uterine adenomyoma has not been reported, to the best of the author’s

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Arch Gynecol Obstet (2015) 291:399–402

knowledge. The present case shows that decidualization occurs in the uterine adenomyoma during pregnancy. Immunohistochemical studies of the decidua have not been reported, to the best of the author’s knowledge. In the present study, the decidua of uterine adenomyoma was positive for pancytokeratin AE1/3, pancytokeratin CAM5.2, CK7, CK18, vimentin, CA125, CD10, estrogen receptor (ER), progesterone receptor (PgR), and Ki-67 (labeling 1 %). They were negative for CK34bE12, CK5/6, CK8, CK14, CK19, CK20, EMA, p63, desmin, a-smooth muscle actin, S100 protein, CK34, CD68, and p53. The positivity of CK AE1/3, CK CAM5.2, CK7 and CK18 in the decidual cells of adenomyoma is curious. In general CKs are expressed in epithelial cells. The expression of CK in decidual cells may indicate that the decidual cells have some epithelial characteristics. The decidual cells were epithelioid in HE staining. This may be related to the positive expression of CKs. The positive vimentin and CD10 are compatible with decidual cells, and indicates that the decidual cells in the present study are mesenchymal cells. CD10 is expressed in normal and neoplastic endometrial stromal cells [21]. The present case showed that CD10 is also expressed in deciduas. The positive expression of CA125, a marker of endometrial glands, in the deciduas is also interesting. This indicates that decidual cells can express CA125. The positive expression of ER and PgR in the deciduas is expected, and indicates that the decidual cells in adenomyoma are controlled by estrogen and progesterone. The low Ki-67 labeling (1 %) in the decidual cells indicates low proliferative fraction. The negative reaction to CK34bE12, CK5/6, CK8, CK14, CK19, CK20, EMA, p63, desmin, a-smooth muscle actin, S100 protein, CK34, CD68 and p53 in the decidual cells indicates that the decidual cells in uterine adenomyoma lack these substances. The immunoprofile of the smooth muscle component in the present adenomyoma is compatible with smooth muscle tumor. The low Ki-67 labeling (1 %) and negative p53 indicate that the present tumor is not leiomyosarcoma. The presence of ER and PgR in the smooth muscle component indicates that the smooth muscles in uterine adenomyoma are influenced by estrogen and progesterone. The immunoprofile of endometrial gland in the present adenomyoma is compatible with normal endometrium [22]. In conclusion, the author, for the first time, reported decidualization of uterine adenomyoma during pregnancy. In addition, some interesting immunohistochemical findings of the deciduas in adenomyoma are reported.

Conflict of interest

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The author has no conflict of interest.

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Decidualization of uterine adenomyoma in a pregnant woman: a case report with immunohistochemical study.

Decidualization of uterine adenomyoma has not been reported, to the best of the author's knowledge...
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