Al Kiswani Somaya, Daas Omar, Al Jabi Mira, Shamiah Mo'ath, Nofal Abdullah, Dibas Yazan
Radiology Department, King Hussein Cancer Center, Amman, Jordan.
Department of Medicine, University of Jordan, Amman, Jordan.
Radiol Case Rep. 2025 Jul 29;20(10):5216-5224. doi: 10.1016/j.radcr.2025.06.116. eCollection 2025 Oct.
Sclerosing epithelioid fibrosarcoma (SEF) is a rare and aggressive variant of fibrosarcoma with a deceptively bland histologic appearance but high metastatic potential, often leading to misdiagnosis and delayed treatment. We present a case of a 21-year-old male initially diagnosed with osteosarcoma due to progressive right proximal thigh pain and swelling. He was treated with perioperative chemotherapy (cisplatin and doxorubicin) and underwent limb-salvage surgery, but postoperative molecular testing revealed an Ewing Sarcoma Receptor 1 (EWSR1) gene rearrangement, confirming SEF. Despite surgery and systemic chemotherapy, the patient developed metastatic disease involving the lungs, adrenal glands, and spine. Aggressive treatments including multiple surgeries, radiotherapy, and chemotherapy (gemcitabine-docetaxel, ifosfamide) failed to halt disease progression. The patient transitioned to palliative care and eventually passed away. SEF is known for its slow progression, high metastatic potential, and resistance to conventional therapies. This case highlights the diagnostic challenges of SEF, the importance of molecular testing for accurate diagnosis, and the need for novel treatment strategies, as SEF remains highly resistant to current therapeutic options.
硬化性上皮样纤维肉瘤(SEF)是纤维肉瘤的一种罕见且侵袭性变体,其组织学表现看似平淡无奇,但转移潜能高,常导致误诊和治疗延误。我们报告一例21岁男性病例,该患者最初因右大腿近端进行性疼痛和肿胀被诊断为骨肉瘤。他接受了围手术期化疗(顺铂和阿霉素)并接受了保肢手术,但术后分子检测显示尤文肉瘤受体1(EWSR1)基因重排,确诊为SEF。尽管进行了手术和全身化疗,患者仍出现了包括肺、肾上腺和脊柱在内的转移病灶。包括多次手术、放疗和化疗(吉西他滨-多西他赛、异环磷酰胺)在内的积极治疗未能阻止疾病进展。患者转为姑息治疗,最终去世。SEF以其进展缓慢、转移潜能高以及对传统疗法耐药而闻名。该病例凸显了SEF的诊断挑战、分子检测对于准确诊断的重要性以及新型治疗策略的必要性,因为SEF对当前的治疗选择仍然高度耐药。