Next-generation sequencing identified an EWSR1::FLI fusion, confirming the diagnosis of an extremely rare primary superficial Ewing sarcoma of the rectum in a 38-year-old male.
Case Report (n=1)
No
This report describes the second molecularly confirmed case of primary superficial Ewing sarcoma of the rectum, highlighting the utility of molecular diagnostics in rare extraosseous presentations.
Ewing sarcoma is an undifferentiated small round cell sarcoma that most commonly presents as a malignant bone tumor in pediatric and young adult patients. The diagnosis is typically confirmed by molecular genetic identification of a fusion protein, most commonly involving members of the FET and ETS gene families. The most frequent translocation is EWSR1::FLI1 which occurs in approximately 85% of cases. Rare extraosseous cases of superficial Ewing sarcoma have been described, including one molecularly confirmed tumor involving the rectum. Herein, we report the second molecularly confirmed case of a superficial, rectal primary tumor arising in a 38-year-old male who initially presented with bleeding. Physical exam noted an ulcerated rectal polyp near the dentate line. Excisional biopsy of the lesion revealed a monotonous round cell malignancy involving the submucosal tissue with necrosis and marked mitotic activity. Immunohistochemical studies showed reactivity for CD99 and NKX2.2 with weak, focal expression of synaptophysin. Next-generation sequencing revealed an EWSR1::FLI fusion, confirming the diagnosis of Ewing sarcoma. The fusion variant junction was located in exon 7 for EWSR1 and exon 6 for FLI1. Subsequent imaging revealed no evidence of metastatic disease, and re-excision showed no residual tumor. Primary superficial Ewing sarcoma is extremely rare, and this is only the second molecularly confirmed case of rectal origin.
Muldoon et al. (2026) conducted a case report in Superficial Ewing Sarcoma of the Rectum (n=1). Excisional biopsy and next-generation sequencing was evaluated. Next-generation sequencing identified an EWSR1::FLI fusion, confirming the diagnosis of an extremely rare primary superficial Ewing sarcoma of the rectum in a 38-year-old male.
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