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Initial biopsies showed bland fibro-osseous lesions lacking GNAS or MDM2 alterations and were favored to represent polyostotic fibrous dysplasia. Subsequent iliac curettage revealed a fascicular spindle cell proliferation with subtle atypia and focal ossification, leading to a revised diagnosis of low-grade fibrosarcoma. Re-biopsy of the T2 lesion demonstrated a similar spindle cell proliferation without ossification. Sequencing identified copy number gains involving KIT, PDGFRA, and TERT. At 16-month follow-up, two metastatic pulmonary nodules developed, one responsive to chemotherapy. The patient remains alive with disease at 27 months after presentation. This case highlights a diagnostic pitfall in which low-grade fibrosarcoma with denosumab-associated ossification may mimic fibrous dysplasia and underscores the prognostic value of molecular profiling beyond histologic grading.<\/jats:p>","DOI":"10.4132\/jptm.2026.05.29","type":"journal-article","created":{"date-parts":[[2026,8,3]],"date-time":"2026-08-03T07:21:02Z","timestamp":1785741662000},"page":"578-586","update-policy":"https:\/\/doi.org\/10.4132\/crossmark_policy","source":"Crossref","is-referenced-by-count":0,"title":["Fibrosarcoma of bone masquerading as fibrous dysplasia: diagnostic pitfall and molecular correlates of aggressive behavior"],"prefix":"10.4132","volume":"60","author":[{"ORCID":"https:\/\/orcid.org\/0000-0002-9538-9535","authenticated-orcid":true,"given":"Wangpan J.","family":"Shi","sequence":"first","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-8087-3327","authenticated-orcid":true,"given":"Brady 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