ArticleHead and neck pathology2025
Rare FUS::FEV-Rearranged Adamantinoma-Like Ewing Sarcoma Arising in the Submandibular Gland.
Article in Head and neck pathology, 2025. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Not yet cited in PubMed.
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Abstract
backgroundAdamantimoma-Like Ewing Sarcoma (ALES) is a rare variant of Ewing sarcoma that poses significant diagnostic challenges in the head and neck due to morphologic overlap with basaloid carcinomas and neuroendocrine neoplasms. CASE PRESENTATION: We report a case of a middle-aged man with a left submandibular gland mass showing classic histologic features of ALES, including basaloid nests with peripheral palisading, rosette formation, and high mitotic activity.
resultsImmunohistochemistry showed co-expression of p63/p40, CD99, and cytokeratins, with patchy synaptophysin positivity and an elevated Ki-67 proliferation index. FISH for EWSR1 was negative, but next-generation sequencing identified a rare FUS::FEV fusion, previously reported only three times in ALES and never in the submandibular gland.
conclusionThis case highlights the importance of extended molecular testing when EWSR1 FISH is negative; as alternate FET::ETS fusions may underlie the diagnosis.
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