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棘状毛发发育异常:病例报告及文献综述

Trichodysplasia spinulosa: Case reports and review of literature.

作者信息

Jose Aju, Dad Taimur, Strand Andrew, Tse Julie Y, Plotnikova Natalia, Boucher Helen W, Sarnak Mark J, Gilbert Scott J, Goyal Nitender

机构信息

Division of Nephrology, Tufts Medical Center, Boston, Massachusetts, USA.

Division of Geographic Medicine and Infectious Diseases, Tufts Medical Center, Boston, Massachusetts, USA.

出版信息

Transpl Infect Dis. 2020 Oct;22(5):e13342. doi: 10.1111/tid.13342. Epub 2020 Jun 16.

Abstract

Trichodysplasia spinulosa (TS) is a rare skin condition caused by trichodysplasia spinulosa-associated polyomavirus (TSPyV). It affects immunosuppressed patients, and <50 cases have been reported. The majority of these cases are seen in solid organ transplant recipients. TS often poses a diagnostic and therapeutic challenge because of its rarity and resemblance with other skin conditions. Several forms of treatment are usually tried prior to establishing a definitive diagnosis. Oral valganciclovir and topical cidofovir have been found to give the best results and hence are the most commonly used agents once the diagnosis is established. Here, we present two cases with a review of literature of TS in solid organ transplant recipients, focusing on time to develop the condition post-transplant, immunosuppression regimen used, and treatment initiated both before and after a definitive diagnosis.

摘要

棘状毛发发育异常(TS)是一种由棘状毛发发育异常相关多瘤病毒(TSPyV)引起的罕见皮肤病。它影响免疫抑制患者,报告的病例不足50例。这些病例大多数见于实体器官移植受者。由于其罕见性以及与其他皮肤病相似,TS常常带来诊断和治疗挑战。在确立明确诊断之前,通常会尝试多种治疗方法。已发现口服缬更昔洛韦和外用西多福韦效果最佳,因此一旦确诊,它们是最常用的药物。在此,我们报告两例病例,并对实体器官移植受者中TS的文献进行综述,重点关注移植后发病时间、所使用的免疫抑制方案以及确诊前后开始的治疗。

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