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汗管瘤样血管角皮瘤合并单发血管角皮瘤或疣状静脉畸形:两例报告及文献复习。

Eccrine angiokeratomatous hamartoma combined with solitary angiokeratoma or verrucous venous malformation: Report of two cases and comprehensive review of the literature.

机构信息

Pathology Unit, S. Orsola Malpighi Hospital, Bologna University, Bologna, Italy.

Dermatology Unit, S. Orsola Malpighi Hospital, Bologna University, Bologna, Italy.

出版信息

J Cutan Pathol. 2020 Jun;47(6):566-570. doi: 10.1111/cup.13654. Epub 2020 Feb 11.

DOI:10.1111/cup.13654
PMID:31997363
Abstract

First reported in 2006, eccrine angiokeratomatous hamartoma is a very rare vascular malformation of the skin, with only few described cases. It has a peculiar histopathology with features deriving from the combination of two different vascular malformations of the skin: solitary angiokeratoma and eccrine angiomatous hamartoma. In the past, other authors described similar hamartomatous lesions with features deriving from verrucous venous malformation and eccrine angiomatous hamartoma. We believe that these lesions are clearly overlapping from clinical, histopathological, and immunohistochemical points of view and the term "eccrine angiokeratomatous hamartoma" should be used to indicate the whole spectrum of these lesions as suggested by Kanitakis et al. Herein we present two cases of this rare vascular hamartoma, with clinical, histopathological and immunohistochemical characterization. In addition, for the first time we report a complete and detailed review of the literature to clarify the clinical, epidemiological, and histopathological features of this unique entity.

摘要

首例报告于 2006 年的小汗腺汗管角化瘤样错构瘤是一种非常罕见的皮肤血管畸形,仅有少数病例报道。其具有独特的组织病理学特征,源于两种不同的皮肤血管畸形:单纯性汗管角化瘤和小汗腺汗管瘤样错构瘤。过去,其他作者曾描述过具有类似错构瘤特征的病变,这些病变源于疣状静脉畸形和小汗腺汗管瘤样错构瘤。我们认为,从临床、组织病理学和免疫组织化学的角度来看,这些病变是明显重叠的,正如 Kanitakis 等人所建议的,应该使用“小汗腺汗管角化瘤样错构瘤”来表示这些病变的全貌。在此,我们介绍了两例这种罕见的血管错构瘤,对其进行了临床、组织病理学和免疫组织化学特征的分析。此外,我们首次对文献进行了全面详细的回顾,以阐明该独特实体的临床、流行病学和组织病理学特征。

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引用本文的文献

1
Intravascular Papillary Endothelial Hyperplasia in Association with Eccrine Angiokeratomatous Hamartoma.血管内乳头状内皮细胞增生伴小汗腺血管角化瘤性错构瘤
Indian J Dermatol. 2023 Nov-Dec;68(6):725. doi: 10.4103/ijd.IJD_189_17. Epub 2024 Jan 9.