Extraskeletal Ewing sarcoma with uterine cornua attachment mimicking high grade endometrial stromal sarcoma: A case report and brief literature review

Background: Ewing sarcoma is an EWSR1-rearranged aggressive malignancy that occurs commonly in bone and has small round blue cell morphology. A diagnostic challenge is presented in the cases of extraskeletal Ewing sarcoma involving solid organs, such as the uterus. Case report and brief literature r...

Full description

Saved in:
Bibliographic Details
Main Authors: Wangpan Shi, Somaye Zare, Cheryl Saenz, Omonigho Aisagbonhi
Format: Article
Language:English
Published: Elsevier 2024-12-01
Series:Gynecologic Oncology Reports
Subjects:
Online Access:http://www.sciencedirect.com/science/article/pii/S2352578924002169
Tags: Add Tag
No Tags, Be the first to tag this record!
_version_ 1850103620853301248
author Wangpan Shi
Somaye Zare
Cheryl Saenz
Omonigho Aisagbonhi
author_facet Wangpan Shi
Somaye Zare
Cheryl Saenz
Omonigho Aisagbonhi
author_sort Wangpan Shi
collection DOAJ
description Background: Ewing sarcoma is an EWSR1-rearranged aggressive malignancy that occurs commonly in bone and has small round blue cell morphology. A diagnostic challenge is presented in the cases of extraskeletal Ewing sarcoma involving solid organs, such as the uterus. Case report and brief literature review: We present the case of a 54-year-old female with a large pelvic mass connected to the uterine cornua and retroperitoneal soft tissue. Upon surgical debulking, the mass was characterized by infiltrative tumor cells with high nuclei-to-cytoplasmic ratio invading from the serosal surface into the myometrium. The initial diagnostic work-up was muddied by radiographic suggestion of uterine origin, histologic identification of uterine involvement and diffuse positivity for Cyclin D1, KIT and CD99, which raised the diagnostic possibility of high-grade endometrial stromal sarcoma. However, molecular testing that revealed EWSR1::FLI fusion, indicative of Ewing sarcoma. She was thus referred from the gynecologic oncology service to the soft tissue sarcoma service for adjuvant chemotherapy of vincristine/doxorubicin/cyclophosphamide and ifosfamide/etoposide (VDC/IE). We performed a literature review highlighting immunohistochemical and molecular features along with treatment and outcomes in case series of YWHAE-rearranged high-grade endometrial stromal sarcomas and Ewing sarcoma with uterine and/or retroperitoneal involvement. Conclusion: This case and accompanying literature review highlight the benefit of molecular testing in the context of round cell tumors involving the uterus and the importance of distinguishing high-grade uterine stromal sarcomas from sarcomas of soft tissue origin, as they have different treatment strategies.
format Article
id doaj-art-89c221fd93c840b0ae774ecbaee7f993
institution DOAJ
issn 2352-5789
language English
publishDate 2024-12-01
publisher Elsevier
record_format Article
series Gynecologic Oncology Reports
spelling doaj-art-89c221fd93c840b0ae774ecbaee7f9932025-08-20T02:39:31ZengElsevierGynecologic Oncology Reports2352-57892024-12-015610153710.1016/j.gore.2024.101537Extraskeletal Ewing sarcoma with uterine cornua attachment mimicking high grade endometrial stromal sarcoma: A case report and brief literature reviewWangpan Shi0Somaye Zare1Cheryl Saenz2Omonigho Aisagbonhi3Department of Pathology, University of California San Diego, La Jolla, CA, USA; Corresponding author.Department of Pathology, University of California San Diego, La Jolla, CA, USAMoores Cancer Center, University of California San Diego, La Jolla, CA, USA; Department of Obstetrics, Gynecology and Reproductive Sciences, University of California San Diego, La Jolla, CA, USADepartment of Pathology, University of California San Diego, La Jolla, CA, USA; Moores Cancer Center, University of California San Diego, La Jolla, CA, USABackground: Ewing sarcoma is an EWSR1-rearranged aggressive malignancy that occurs commonly in bone and has small round blue cell morphology. A diagnostic challenge is presented in the cases of extraskeletal Ewing sarcoma involving solid organs, such as the uterus. Case report and brief literature review: We present the case of a 54-year-old female with a large pelvic mass connected to the uterine cornua and retroperitoneal soft tissue. Upon surgical debulking, the mass was characterized by infiltrative tumor cells with high nuclei-to-cytoplasmic ratio invading from the serosal surface into the myometrium. The initial diagnostic work-up was muddied by radiographic suggestion of uterine origin, histologic identification of uterine involvement and diffuse positivity for Cyclin D1, KIT and CD99, which raised the diagnostic possibility of high-grade endometrial stromal sarcoma. However, molecular testing that revealed EWSR1::FLI fusion, indicative of Ewing sarcoma. She was thus referred from the gynecologic oncology service to the soft tissue sarcoma service for adjuvant chemotherapy of vincristine/doxorubicin/cyclophosphamide and ifosfamide/etoposide (VDC/IE). We performed a literature review highlighting immunohistochemical and molecular features along with treatment and outcomes in case series of YWHAE-rearranged high-grade endometrial stromal sarcomas and Ewing sarcoma with uterine and/or retroperitoneal involvement. Conclusion: This case and accompanying literature review highlight the benefit of molecular testing in the context of round cell tumors involving the uterus and the importance of distinguishing high-grade uterine stromal sarcomas from sarcomas of soft tissue origin, as they have different treatment strategies.http://www.sciencedirect.com/science/article/pii/S2352578924002169EwingUterusEndometrialStromalSarcomaCancer
spellingShingle Wangpan Shi
Somaye Zare
Cheryl Saenz
Omonigho Aisagbonhi
Extraskeletal Ewing sarcoma with uterine cornua attachment mimicking high grade endometrial stromal sarcoma: A case report and brief literature review
Gynecologic Oncology Reports
Ewing
Uterus
Endometrial
Stromal
Sarcoma
Cancer
title Extraskeletal Ewing sarcoma with uterine cornua attachment mimicking high grade endometrial stromal sarcoma: A case report and brief literature review
title_full Extraskeletal Ewing sarcoma with uterine cornua attachment mimicking high grade endometrial stromal sarcoma: A case report and brief literature review
title_fullStr Extraskeletal Ewing sarcoma with uterine cornua attachment mimicking high grade endometrial stromal sarcoma: A case report and brief literature review
title_full_unstemmed Extraskeletal Ewing sarcoma with uterine cornua attachment mimicking high grade endometrial stromal sarcoma: A case report and brief literature review
title_short Extraskeletal Ewing sarcoma with uterine cornua attachment mimicking high grade endometrial stromal sarcoma: A case report and brief literature review
title_sort extraskeletal ewing sarcoma with uterine cornua attachment mimicking high grade endometrial stromal sarcoma a case report and brief literature review
topic Ewing
Uterus
Endometrial
Stromal
Sarcoma
Cancer
url http://www.sciencedirect.com/science/article/pii/S2352578924002169
work_keys_str_mv AT wangpanshi extraskeletalewingsarcomawithuterinecornuaattachmentmimickinghighgradeendometrialstromalsarcomaacasereportandbriefliteraturereview
AT somayezare extraskeletalewingsarcomawithuterinecornuaattachmentmimickinghighgradeendometrialstromalsarcomaacasereportandbriefliteraturereview
AT cherylsaenz extraskeletalewingsarcomawithuterinecornuaattachmentmimickinghighgradeendometrialstromalsarcomaacasereportandbriefliteraturereview
AT omonighoaisagbonhi extraskeletalewingsarcomawithuterinecornuaattachmentmimickinghighgradeendometrialstromalsarcomaacasereportandbriefliteraturereview