Department of Pathology and Laboratory Medicine, Nationwide Children's Hospital, Columbus, Ohio, USA.
Department of Pathology, The Ohio State University, Columbus, Ohio, USA.
Genes Chromosomes Cancer. 2021 Sep;60(9):640-646. doi: 10.1002/gcc.22973. Epub 2021 Jun 7.
Gastroblastomas are rare tumors with a biphasic epithelioid/spindle cell morphology that typically present in early adulthood and have recurrent MALAT1-GLI1 fusions. We describe an adolescent patient with Wiskott-Aldrich syndrome who presented with a large submucosal gastric tumor with biphasic morphology. Despite histologic features consistent with gastroblastoma, a MALAT1-GLI1 fusion was not found in this patient's tumor; instead, comprehensive molecular profiling identified a novel EWSR1-CTBP1 fusion and no other significant genetic alterations. The tumor also overexpressed NOTCH and FGFR by RNA profiling. The novel fusion and expression profile suggest a role for epithelial-mesenchymal transition in this tumor, with potential implications for the pathogenesis of biphasic gastric tumors such as gastroblastoma.
胃胚细胞瘤是一种罕见的肿瘤,具有双相上皮样/梭形细胞形态,通常发生在成年早期,并存在反复的 MALAT1-GLI1 融合。我们描述了一位患有 Wiskott-Aldrich 综合征的青少年患者,他患有大的黏膜下胃肿瘤,具有双相形态。尽管组织学特征与胃胚细胞瘤一致,但在该患者的肿瘤中未发现 MALAT1-GLI1 融合;相反,全面的分子分析确定了一种新的 EWSR1-CTBP1 融合,并且没有其他显著的遗传改变。肿瘤还通过 RNA 分析过表达 NOTCH 和 FGFR。新型融合和表达谱提示上皮-间充质转化在该肿瘤中的作用,这可能对双相胃肿瘤(如胃胚细胞瘤)的发病机制有影响。