Open Access Case report

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

Giacomo Puppa1, Makio Shozu2, Tiziana Perin1, Kazuhito Nomura3, Annunziata Gloghini4, Elio Campagnutta5 and Vincenzo Canzonieri1*

Author Affiliations

1 Division of Pathology, Centro di Riferimento Oncologico, Istituto Nazionale Tumori – IRCCS, Via Pedemontana Occidentale 12, 33081 Aviano PN, Italy

2 Graduate School of Chiba University, Reproductive Medicine, Chuo-ku, Chiba, Japan

3 Department of Obstetrics and Gynecology, Kanazawa University School of Medicine, Takara-machi, Kanazawa, Japan

4 Diagnostic Immunohistochemistry and Molecular Pathology Unit, Istituto Nazionale Tumori – IRCCS, Aviano PN, Italy

5 Division of Gynecologic Oncology, Centro di Riferimento Oncologico, Istituto Nazionale Tumori – IRCCS, Aviano PN, Italy

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BMC Cancer 2007, 7:103  doi:10.1186/1471-2407-7-103

Published: 20 June 2007



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.


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