Primary Extra-Uterine Endometrial Stromal Sarcoma and Synchronous Breast Cancer: A Double Whammy: A Case report

Pavneet Kohli, Prasanth Penumadu, Neelesh Srivastava, Bheemanathi Hanuman Srinivas, Vidyalakshmi Rangarajan
Journal of obstetrics and gynaecology of India · 2021 · vol. 71(3) , pp. 326–329 · doi:10.1007/s13224-021-01437-6 · PMID:34408355 · PMC8310824 · W3126431946
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This case report describes a rare instance of a 40-year-old woman diagnosed with both extra-uterine endometrial stromal sarcoma and synchronous invasive ductal breast carcinoma.

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This case report describes a 40-year-old woman with bilateral adnexal tumors whose diagnostic workup revealed extra-uterine endometrial stromal sarcoma (EESS) and a synchronous invasive ductal breast carcinoma that was hormone-positive. The authors highlight that histopathology confirmed EESS in the absence of florid endometriosis, while the breast malignancy represented synchronous hormone receptor–positive infiltrating duct cancer, creating a diagnostic challenge. The patient received adjuvant endocrine therapy and was reported disease-free at 2 years, with the limitation inherent to a single-patient case report and brief follow-up. Relevance to endometriosis: the paper explicitly notes EESS occurred “in the absence of florid endometriosis,” which directly bears on endometriosis-associated differential diagnosis and symptom/pathology overlap, though the main focus is the unusual synchronous presentation of EESS and breast cancer.

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Abstract

Endometrial stromal sarcoma is an uncommon gynecological tumor. Extra-uterine endometrial stromal sarcoma (EESS), the extra-uterine variant of its relatively more common counterpart, is even rarer with only few documented case reports. We report a case of a 40-year woman with bilateral adnexal tumors and synchronous invasive ductal breast carcinoma (IDC) posing a diagnostic challenge. The histopathology of specimen confirmed the diagnosis of EESS in the absence of florid endometriosis and synchronous hormone-positive infiltrating duct cancer in the breast. Patient was started on adjuvant endocrine therapy and is disease free at the end of 2 years. To the best of our knowledge, this is the first documentation of synchronous presentation of IDC and EESS, highlighting the possible role of hyper-estrogenemia as an etiological factor.

References

Masand RP, Euscher ED, Deavers MT, et al. Endometrioid stromal sarcoma. Am J Surg Pathol. 2013;37(11):1635–47. https://doi.org/10.1097/PAS.0000000000000083. Tian W, Latour M, Epstein JI. Endometrial stromal sarcoma involving the urinary bladder: a study of 6 cases. Am J Surg Pathol. 2014;38(7):982–9. https://doi.org/10.1097/PAS.0000000000000214. Oliva E, Egger JF, Young RH. A clinicopathologic study of 27 cases with morphologic and behavioral features similar to those of uterine low-grade endometrial stromal sarcoma. Am J Surg Pathol. 2014;38(3):305–15. https://doi.org/10.1097/PAS.0000000000000145. Altman AD, Nelson GS, Chu P, et al. Uterine sarcoma and aromatase inhibitors: Tom Baker cancer centre experience and review of the literature. Int J Gynecol Cancer. 2012;22(6):1006–12. https://doi.org/10.1097/IGC.0b013e31825b7de8. Funding No funding or financial support was received from any commercial organization. Authors declare no conflicts of interest with anybody Author information Authors and Affiliations Corresponding author Additional information Publisher's Note Springer Nature remains neutral with regard to jurisdictional claims in published maps and institutional affiliations. Dr. Pavneet Kohli is an Assistant professor, Department of Surgical Oncology, JIPMER, Puducherry, India; Dr. Prasanth Penumadu is an Associate professor, Department of Surgical Oncology, JIPMER, Puducherry, India; Dr. Neelesh Srivastava is a senior resident, Department of Surgical Oncology, JIPMER, Puducherry, India; Dr. Bheemanathi Hanuman Srinivas is an Additional Professor, Department of Pathology, JIPMER, Puducherry, India; Dr. Vidyalakshmi Rangarajan is a senior resident, Department of Pathology, JIPMER, Puducherry, India. Rights and permissions About this article Cite this article Kohli, P., Penumadu, P., Srivastava, N. et al. Primary Extra-Uterine Endometrial Stromal Sarcoma and Synchronous Breast Cancer: A Double Whammy: A Case report. J Obstet Gynecol India 71, 326–329 (2021). https://doi.org/10.1007/s13224-021-01437-6 Received: Accepted: Published: Version of record: Issue date: DOI: https://doi.org/10.1007/s13224-021-01437-6

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