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免疫抑制相关的病毒相关性毛发发育异常:1例口服缬更昔洛韦治疗有效的儿科患者报告

Viral-associated trichodysplasia of immunosuppression: report of a pediatric patient with response to oral valganciclovir.

作者信息

Benoit Taylor, Bacelieri Rocky, Morrell Dean S, Metcalf John

机构信息

Department of Dermatology and Dermatologic Surgery, Medical University of South Carolina, 135 Rutledge Ave., Charleston, SC 29425, USA.

出版信息

Arch Dermatol. 2010 Aug;146(8):871-4. doi: 10.1001/archdermatol.2010.175.

Abstract

BACKGROUND

Viral-associated trichodysplasia of immunosuppression is an increasingly recognized entity characterized by follicular-based papules, primarily in the central part of the face, that produce variable degrees of alopecia and dysmorphic features. It has been primarily described in transplant recipients but has recently been recognized in patients receiving chemotherapy for leukemia and lymphoma. It is associated with distinctive histologic features such as dilated anagen hair follicles, absent hair papillae, and abrupt cornification of the inner root sheath.

OBSERVATIONS

A 5-year-old boy presented with spiny follicular papules that caused thickening of the skin of the face 1 year after cardiac transplantation. He had been exposed to several immunosuppressive agents, including mycophenolate mofetil, tacrolimus, intravenous immunoglobulin, rituximab, cylcophosphamide, and prednisone. Despite the failure of multiple topical treatments, our patient's eruption improved with systemic valganciclovir therapy.

CONCLUSIONS

We describe the youngest patient (to our knowledge) with viral-associated trichodysplasia of immunosuppression and discuss the characteristic clinicopathologic features. Our report supports the theory that immunosuppression is the predisposing factor to a folliculotropic papovavirus that alters follicular maturation.

摘要

背景

免疫抑制相关的病毒性毛发发育异常是一种越来越被认识到的疾病,其特征为以毛囊为基础的丘疹,主要位于面部中央,可导致不同程度的脱发和畸形特征。该病主要在移植受者中被描述,但最近在接受白血病和淋巴瘤化疗的患者中也有发现。它与独特的组织学特征相关,如生长期毛囊扩张、毛乳头缺失以及内根鞘突然角化。

观察结果

一名5岁男孩在心脏移植1年后出现棘状毛囊丘疹,导致面部皮肤增厚。他曾接触多种免疫抑制剂,包括霉酚酸酯、他克莫司、静脉注射免疫球蛋白、利妥昔单抗、环磷酰胺和泼尼松。尽管多种局部治疗均无效,但我们的患者经全身使用缬更昔洛韦治疗后皮疹有所改善。

结论

我们描述了(据我们所知)最年轻的免疫抑制相关的病毒性毛发发育异常患者,并讨论了其特征性的临床病理特征。我们的报告支持免疫抑制是易感染向毛囊趋化的乳头多瘤空泡病毒从而改变毛囊成熟的 predisposing 因素这一理论。 (注:predisposing 这个词在医学语境中不太好准确翻译,大概意思是“使易患的、诱发的”,这里直接保留英文供参考)

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