Department of Hepatopancreatobiliary Surgery, Chongqing General Hospital, Chongqing, China (mainland).
Institute of Pathology and Southwest Cancer Center, Southwest Hospital, Army Medical University, Chongqing, China (mainland).
Am J Case Rep. 2024 May 23;25:e943271. doi: 10.12659/AJCR.943271.
BACKGROUND Soft tissue tumors have various subtypes, among which sarcomas exhibit high malignant potential and poor prognosis. Malignant epithelioid tumor with GLI1 alterations was originally found in myopericytoma with t(7;12) translocation. However, recent studies indicated that it is a distinct tumor type characterized by multiple nodular distributions of oval or round epithelioid cells with a rich capillary network and a lack of specific immunophenotype. There are only a few cases reported worldwide and the optimal treatment is still being explored. CASE REPORT We report the case of a 31-year-old patient who presented with severe anemia and a large soft tissue mass in the duodenum. The patient underwent surgical resection with a negative margin, and none of the 15 lymph nodes tested positive for the tumor. Postoperative pathology and FISH testing further confirmed the presence of GLI1 disruption and S-100 and SMA negativity. Genetic testing revealed the ACTB-GLI1 fusion. No specific medication was offered after the surgery. No tumor recurrence was found during the 23-month follow-up period. The patient's quality of life is currently satisfactory. CONCLUSIONS Soft tissue sarcomas characterized by GLI1 gene rearrangement have a relatively less aggressive and metastatic nature, with the solid mass spreading minimally even as it grows. Patients can benefit from surgical resection, resulting in a relatively long period of tumor-free survival.
软组织肿瘤有多种亚型,其中肉瘤具有较高的恶性潜能和较差的预后。具有 GLI1 改变的恶性上皮样肿瘤最初在具有 t(7;12)易位的肌纤维母细胞瘤中发现。然而,最近的研究表明,它是一种独特的肿瘤类型,其特征是多个圆形或椭圆形上皮样细胞结节分布,富含毛细血管网络,缺乏特异性免疫表型。目前全世界仅有少数病例报道,最佳治疗方法仍在探索中。
我们报告了一例 31 岁患者,其表现为严重贫血和十二指肠内大的软组织肿块。患者行手术切除,切缘阴性,15 枚送检淋巴结均未检出肿瘤。术后病理及 FISH 检测进一步证实存在 GLI1 断裂和 S-100、SMA 阴性。基因检测显示 ACTB-GLI1 融合。术后未提供特异性药物治疗。23 个月的随访期间未发现肿瘤复发。患者目前生活质量满意。
具有 GLI1 基因重排的软组织肉瘤具有相对侵袭性和转移性较弱的特点,即使在生长过程中,实体瘤的扩散也很小。患者受益于手术切除,从而获得相对较长的无瘤生存期。