El-Sharkawy Farah, Delgado Patricia Isabel, Podda Antonello, Neville Holly Leigh, Rojas Claudia Patricia
1 Department of Pathology, University of Miami Health System/Jackson Memorial Hospital, Miami, Florida, USA.
2 Department of Pediatric Hematology-Oncology, University of Miami Health System/Jackson Memorial Hospital, Miami, Florida, USA.
Pediatr Dev Pathol. 2017 Mar-Apr;20(2):163-167. doi: 10.1177/1093526616686007. Epub 2017 Jan 26.
Angiosarcomas are highly aggressive malignancies of vascular origin and are very rarely found in children. We report a case of a 13-year-old girl with a history of abdominal pain and increased abdominal girth. Radiologic imaging showed significant ascites and large pelvic masses involving bilateral adnexa with abdominal spread. Microscopic examination of a biopsy revealed pleomorphic epithelioid and spindle cells with brisk mitotic activity, intracytoplasmic vacuoles, vascular channels, and large areas of hemorrhage and necrosis. Immunohistochemistry analysis showed strong and diffuse positivity for CD31, D2-40, ERG, FLI-1, and focally for CD34, vWF, and EMA. The diagnosis of metastatic angiosarcoma was rendered. The patient was treated aggressively with systemic chemotherapy, immunotherapy, cytoreductive surgery, and hyperthermic intraperitoneal chemotherapy, with a favorable response after 1-year follow-up. Angiosarcoma should be considered when encountering a vascular tumor with pleomorphism, brisk mitotic activity, and necrosis. Immunohistochemistry studies are necessary for proper diagnosis.
血管肉瘤是一种起源于血管的高度侵袭性恶性肿瘤,在儿童中极为罕见。我们报告一例13岁女孩,有腹痛和腹围增加病史。放射影像学检查显示大量腹水和累及双侧附件并伴有腹腔播散的巨大盆腔肿块。活检组织显微镜检查显示多形性上皮样细胞和梭形细胞,有活跃的有丝分裂活性、胞质内空泡、血管腔隙以及大片出血和坏死。免疫组织化学分析显示CD31、D2-40、ERG、FLI-1呈强弥漫性阳性,CD34、vWF和EMA呈局灶性阳性。诊断为转移性血管肉瘤。该患者接受了全身化疗、免疫治疗、减瘤手术和腹腔内热灌注化疗等积极治疗,随访1年后反应良好。遇到具有多形性、活跃有丝分裂活性和坏死的血管肿瘤时应考虑血管肉瘤。免疫组织化学研究对于正确诊断是必要的。