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头颈部骨内乳头状血管瘤 1 例罕见报告

A Rare Case of Intraosseous Papillary Hemangioma of the Head and Neck.

机构信息

Department of Pathology and Laboratory Medicine, North Shore University Hospital and Long Island Jewish Medical Center, Donald and Barbara Zucker School of Medicine at Hofstra/Northwell Health, Greenvale, NY, USA.

Department of Pathology, Northern Westchester and Phelps Hospital, Donald and Barbara Zucker School of Medicine at Hofstra/Northwell Health, Greenvale, NY, USA.

出版信息

Int J Surg Pathol. 2024 Apr;32(2):418-421. doi: 10.1177/10668969231180282. Epub 2023 Jun 12.

Abstract

Papillary hemangioma is a novel variant of intravascular hemangioma. It is more common in adults and has a male predominance. Most tumors reported so far are solitary and cutaneous. Here we present a rare case of an intraosseous papillary hemangioma involving the frontal bone. Brain imaging in a 69-year-old man with a slowly enlarging swelling on the right frontal area following an accidental fall demonstrated a 4.5 cm × 1.7 cm × 4.2 cm mass originating from the right frontal bone, with a tiny defect on the orbital roof. A malignant process was favored, and the mass was removed. Histopathology revealed a vascular lesion showing intraosseous distribution with foci of extension into the fibrous connective tissue. There were areas of plump endothelial cells with intracytoplasmic hyaline globules arranged in papillary configuration. The lesional cells were immunoreactive with CD34. AE1/AE3, EMA, PR, D2-40, inhibin, and S100 stains were negative. Ki-67 was low. This is the first intraosseous and second noncutaneous papillary hemangioma. Clinically it differs from other cases by the presence of trauma as a preceding event. Since its prognosis is unknown such patients should be monitored for recurrence or malignant transformation.

摘要

乳头状血管瘤是一种新型的血管内血管瘤。它在成年人中更为常见,且男性居多。迄今为止报道的大多数肿瘤都是单发的皮肤性肿瘤。在此,我们报告一例罕见的额骨内骨内乳头状血管瘤。一位 69 岁男性因右侧额部意外摔伤后逐渐增大的肿块就诊,头部影像学检查显示一个源于右侧额骨的 4.5cm×1.7cm×4.2cm 的肿块,眶顶有一个微小的缺损。考虑为恶性病变,于是进行了肿块切除术。组织病理学显示,血管病变呈骨内分布,局部向纤维结缔组织延伸。有一些细胞胞质内含有嗜酸性透明小体,呈乳头状排列的饱满内皮细胞区。病变细胞 CD34 阳性,AE1/AE3、EMA、PR、D2-40、抑制素和 S100 染色均为阴性,Ki-67 低表达。这是首例骨内和第二例非皮肤的乳头状血管瘤。与其他病例不同,该例患者存在创伤这一先前事件。由于其预后尚不清楚,此类患者应密切监测复发或恶性转化的情况。

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