Kim Gilhyang, Kim Jin Cheol, Kang Seok Young, Kim Hye One, Park Chun Wook, Chung Bo Young
Department of Pathology, Hallym University Kangnam Sacred Heart Hospital, College of Medicine, Hallym University, Seoul, Korea.
Department of Dermatology, Hallym University Kangnam Sacred Heart Hospital, College of Medicine, Hallym University, Seoul, Korea.
Ann Dermatol. 2023 Nov;35(Suppl 2):S252-S255. doi: 10.5021/ad.21.221.
Epithelioid hemangioma is a rare benign vascular neoplasm. Recently, the term "multiple eruptive epithelioid hemangioma" has been proposed for epithelioid hemangioma with distinct features. It is different from usual epithelioid hemangioma because of the multifocal distributions in various body regions with an eruptive onset. In addition, the histopathology of multiple eruptive epithelioid hemangioma shows increased cellular proliferation, mitosis, and nuclear pleomorphism and positive findings for FOS-B compared to classic epithelioid hemangioma. Herein, we report the case of a 59-year-old man with unusual manifestations suitable for multiple eruptive epithelioid hemangioma. He had multiple erythematous to purple-red dome-shaped nodules on the right hand, arm, and shoulder. The initial lesion was a solitary erythematous nodule on the right hand that abruptly extended to the right arm and shoulder. Microscopically, the tumor was a well-demarcated dermal nodule and showed capillary sized vascular structures. Vascular structures had epithelioid endothelial cells with abundant eosinophilic cytoplasm and vesicular nuclei. The tumor cells showed mild nuclear pleomorphism and a few mitosis and feature of resembling cobble stone was observed. In immunohistochemistry, CD31 and CD34 were positive in the endothelial cells. The endothelial cells showed nuclear positivity in FOS-B. Based on the clinical and histopathological findings, the final diagnosis was multiple eruptive epithelioid hemangiomas. This is the first report of multiple eruptive epithelioid hemangiomas in an Asian man after the term had been introduced.
上皮样血管瘤是一种罕见的良性血管肿瘤。最近,“多发性暴发性上皮样血管瘤”这一术语被用于描述具有独特特征的上皮样血管瘤。它与普通上皮样血管瘤不同,因为其在身体各个部位多灶性分布且起病急骤。此外,与经典上皮样血管瘤相比,多发性暴发性上皮样血管瘤的组织病理学表现为细胞增殖增加、有丝分裂和核多形性增加,且FOS - B呈阳性。在此,我们报告一例59岁男性,其临床表现不寻常,符合多发性暴发性上皮样血管瘤。他的右手、手臂和肩部有多个从红斑到紫红色的圆顶状结节。最初的病变是右手的一个孤立性红斑结节,随后突然扩展到右臂和肩部。显微镜下,肿瘤是一个边界清晰的真皮结节,可见毛细血管大小的血管结构。血管结构有上皮样内皮细胞,胞质丰富嗜酸性,核呈泡状。肿瘤细胞显示轻度核多形性,有少量有丝分裂,可见类似鹅卵石的特征。免疫组化显示,内皮细胞中CD31和CD34呈阳性。内皮细胞FOS - B呈核阳性。根据临床和组织病理学检查结果,最终诊断为多发性暴发性上皮样血管瘤。这是该术语提出后亚洲男性多发性暴发性上皮样血管瘤的首例报告。