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移植物抗宿主病相关血管瘤病:一种临床病理特征独特的疾病实体。

Graft-versus-host disease-associated angiomatosis: a clinicopathologically distinct entity.

作者信息

Kaffenberger Benjamin H, Zuo Rena C, Gru Alejandro, Plotner Alisha N, Sweeney Sarah A, Devine Steven M, Hymes Sharon R, Cowen Edward W

机构信息

Division of Dermatology, Ohio State University Wexner Medical Center, Columbus, Ohio.

Dermatology Branch, Center for Cancer Research, National Cancer Institute, National Institutes of Health, Bethesda, Maryland.

出版信息

J Am Acad Dermatol. 2014 Oct;71(4):745-53. doi: 10.1016/j.jaad.2014.05.034. Epub 2014 Jul 1.

Abstract

BACKGROUND

Chronic graft-versus-host disease (GVHD) may present with various cutaneous manifestations. Isolated case reports describe eruptive angiomas in this setting.

OBJECTIVE

We sought to provide a clinical and pathologic description of vascular proliferations in patients with GVHD.

METHODS

Cases of documented GVHD associated with vascular proliferations were collected from the National Institutes of Health, Ohio State University, and MD Anderson Cancer Center.

RESULTS

Eleven patients with a diagnosis of GVHD who developed vascular proliferations were identified. All patients manifested sclerotic type chronic GVHD of the skin. Vascular lesions were first documented a median of 44 months after transplantation and occurred primarily on the lower extremities or trunk. Histopathology revealed anastomosing networks of thin-walled vascular proliferations in a vague lobular growth pattern, with overlying epidermal acanthosis, peripheral collarette, ulceration, and disorganized fibroblast-rich and fibrotic stroma. Improvement was noted in 1 patient treated with propranolol and sirolimus and 1 patient with electrocautery.

LIMITATIONS

Given the retrospective nature of the study, the overall incidence of vascular lesions in patients with GVHD is unknown. Histopathology was present for review on only 3 of 11 patients.

CONCLUSION

The phenomenon of vascular lesions appears to be relatively specific for sclerotic type chronic GVHD when compared with other fibrosing diseases. We propose the term "graft-versus-host disease-associated angiomatosis" to describe this entity.

摘要

背景

慢性移植物抗宿主病(GVHD)可能表现出多种皮肤表现。孤立病例报告描述了在这种情况下出现的暴发性血管瘤。

目的

我们试图提供GVHD患者血管增生的临床和病理描述。

方法

从美国国立卫生研究院、俄亥俄州立大学和MD安德森癌症中心收集记录有GVHD相关血管增生的病例。

结果

确定了11例诊断为GVHD且发生血管增生的患者。所有患者均表现为皮肤硬化型慢性GVHD。血管病变首次记录于移植后中位数44个月,主要发生在下肢或躯干。组织病理学显示薄壁血管增生呈模糊的小叶状生长模式,形成吻合网络,伴有表皮棘层增厚、周边领圈样改变、溃疡,以及富含成纤维细胞且纤维化的间质紊乱。1例接受普萘洛尔和西罗莫司治疗的患者以及1例接受电灼治疗的患者病情有所改善。

局限性

鉴于该研究的回顾性性质,GVHD患者血管病变的总体发生率未知。11例患者中仅有3例有组织病理学检查结果可供审查。

结论

与其他纤维化疾病相比,血管病变现象似乎对硬化型慢性GVHD具有相对特异性。我们建议用“移植物抗宿主病相关血管瘤病”这一术语来描述这一实体。

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