Small primary adenocarcinoma in adenomyosis with nodal metastasis: a case report

Abstract

Background: Malignant transformation of adenomyosis is a very rare event. Only about 30 cases of this occurrence have been documented till now.

Case presentation: The patient was a 57-year-old woman with a slightly enlarged uterus, who underwent total hysterectomy and unilateral adnexectomy. On gross inspection, the uterine wall displayed a single nodule measuring 5 cm and several small gelatinous lesions. Microscopic examination revealed a common leiomyoma and multiple adenomyotic foci. A few of these glands were transformed into a moderately differentiated adenocarcinoma. The endometrium was completely examined and tumor free. The carcinoma was, therefore, considered to be an endometrioid adenocarcinoma arising from adenomyosis. Four months later, an ultrasound scan revealed enlarged pelvic lymph nodes: a cytological diagnosis of metastatic adenocarcinoma was made. Immunohistochemical studies showed an enhanced positivity of the tumor site together with the neighbouring adenomyotic foci for estrogen receptors, aromatase, p53 and COX-2 expression when compared to the distant adenomyotic glands and the endometrium. We therefore postulate that the neoplastic transformation of adenomyosis implies an early carcinogenic event involving p53 and COX-2; further tumor growth is sustained by an autocrine-paracrine loop, based on a modulation of hormone receptors as well as aromatase and COX-2 local expression.

Conclusion: Adenocarcinoma in adenomyosis may be affected by local hormonal influence and, despite its small size, may metastasize.

Figure 1

(A) The tumor arises from the confluence of transformed foci. (B) Higher power magnification of the tumor. (C) Cell block cytology of the lymph node metastasis. (D) Immunocytochemical staining of the metastasis for CA125.

Figure 2

(A) Diffuse strong positivity for ER of the tumor. (B) Patchy positivity for PR of the tumor. (C) Aromatase positivity of benign adenomyotic foci (arrows). Most neoplastic cells are negative for aromatase (asterisk) except for some glands (insert). (D) COX-2 positivity of the tumor and adenomyosis. (E) CA125 positivity of the tumor and of the adenomyotic epithelium. (F) Focal positivity for p53 of the tumor.

Figure 3

(A) The atrophic and proliferative component in the endometrium, respectively negative and positive for ER. (B) Aromatase positivity in the superficial layer of a part of the endometrium. (C) The adenomyotic glands throughout the uterus: aromatase expressed by some glands (left), whereas other glands are negative (right) (D) p53 positivity in some adenomyotic glands.