Primary endometrioid carcinoma of the uterosacral ligament arising from deep infiltrating endometriosis 6 years after bilateral salpingo-oophorectomy due to atypical proliferative endometrioid tumor of the ovary: a rare case report

Yoshiaki Ota, Kuniaki Ota, Toshifumi Takahashi, Soichiro Suzki, Rikiya Sano, Ikuko Ota, Takuya Moriya, Mitsuru Shiota
World journal of surgical oncology · 2020 · vol. 18(1) , pp. 329 · doi:10.1186/s12957-020-02105-1 · PMID:33308243 · W3110733070
article OA: gold CC0 ⤵ 8 in-corpus citations
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⚙ AI-generated summary by claude@2026-06, 2026-06-07 ⓘ

This case report describes a rare instance of primary endometrioid carcinoma developing in the uterosacral ligament from deep infiltrating endometriosis six years after treatment for a borderline ovarian tumor.

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⚙ AI-generated deep summary by claude@2026-06, 2026-06-07 ⓘ

This paper reports a rare case in a 48-year-old woman with severe pelvic pain who underwent laparoscopic bilateral salpingo-oophorectomy for a borderline atypical proliferative endometrioid tumor of the right ovary and an ovarian endometrioma, with a residual deep infiltrating endometriosis–suspected lesion in the left uterosacral ligament. Six years later, imaging showed an FDG-avid solid tumor in the cul-de-sac, leading to diagnostic laparoscopy followed by hysterectomy, partial omentectomy, and pelvic and para-aortic lymphadenectomy; histology identified well-differentiated endometrioid carcinoma arising from deep infiltrating endometriosis at the uterosacral ligament edge, with a final diagnosis of primary stage IIB peritoneal carcinoma, and the authors note no evidence of recurrence after 2 years post–adjuvant paclitaxel/carboplatin. As a single case report, the main limitation is that it cannot establish incidence, causality, or generalizable treatment effects. This paper is centrally about endometriosis — specifically endometrioid carcinoma developing from deep infiltrating endometriosis in the uterosacral ligament after prior surgery.

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Abstract

Abstract Background Endometriosis can potentially lead to the development of a malignant tumor. Most malignant tumors arising from the endometriosis originate from the ovarian endometrioma, whereas those arising from extragonadal lesions are rare. We report a rare case of endometrioid carcinoma that developed from deep infiltrating endometriosis in the uterosacral ligament 6 years after treatment for atypical proliferative endometrioid tumor of the ovary in a 48-year-old woman. Case presentation Six years ago, the patient underwent laparoscopic bilateral salpingo-oophorectomy for her right ovarian tumor with atypical proliferative (borderline) endometrioid tumor accompanied by ovarian endometrioma. The solid tumor in the cul-de-sac was detected during follow-up using magnetic resonance imaging. Positron emission tomography/computed tomography revealed an abnormal accumulation of 18 F-fluorodeoxyglucose at the tumor site. Thus, tumor recurrence with borderline malignancy was suspected. The patient underwent diagnostic laparoscopy followed by hysterectomy and partial omentectomy. Retroperitoneal pelvic lymphadenectomy and para-aortic lymphadenectomy were also performed. The cul-de-sac tumor at the left uterosacral ligament was microscopically diagnosed as invasive endometrioid carcinoma arising from deep infiltrating endometriosis. The final diagnosis was primary stage IIB peritoneal carcinoma. The patient received six courses of monthly paclitaxel and carboplatin as adjuvant chemotherapy. The patient showed no evidence of recurrence for 2 years after the treatments. Conclusion This study reports a rare case of metachronous endometriosis-related malignancy that developed 6 years after treatment for borderline ovarian tumor. If endometriosis lesions remain after bilateral salpingo-oophorectomy, the physician should keep the malignant nature of endometriosis in mind.

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Condition tags

mesh:D004715endometriosisdie_deep_infiltratingendometrioma

MeSH descriptors

Carcinoma, Endometrioid Carcinoma, Endometrioid Carcinoma, Endometrioid Endometriosis Endometriosis Ovarian Neoplasms Ovarian Neoplasms Ovarian Neoplasms Female Humans Ligaments Middle Aged Neoplasm Recurrence, Local Prognosis Salpingo-oophorectomy

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