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成人免疫缺陷相关风疹病毒感染致肉芽肿性皮炎

Granulomatous Dermatitis Associated With Rubella Virus Infection in an Adult With Immunodeficiency.

机构信息

Department of Dermatology, University of Wisconsin School of Medicine and Public Health, Madison.

Assistant Section Editor, JAMA Dermatology.

出版信息

JAMA Dermatol. 2021 Jul 1;157(7):842-847. doi: 10.1001/jamadermatol.2021.1577.

Abstract

IMPORTANCE

Immunodeficiency-related, vaccine-derived rubella virus (RuV) as an antigenic trigger of cutaneous and visceral granulomas is a rare, recently described phenomenon in children and young adults treated with immunosuppressant agents.

OBJECTIVE

To perform a comprehensive clinical, histologic, immunologic, molecular, and genomic evaluation to elucidate the potential cause of an adult patient's atypical cutaneous granulomas.

DESIGN, SETTING, AND PARTICIPANTS: A prospective evaluation of skin biopsies, nasopharyngeal swabs, and serum samples submitted to the Centers for Disease Control and Prevention was conducted to assess for RuV using real-time reverse-transcriptase polymerase chain reaction (RT-PCR) and viral genomic sequencing. The samples were obtained from a man in his 70s with extensive cutaneous granulomas mimicking both cutaneous sarcoidosis (clinically) and CD8+ granulomatous cutaneous T-cell lymphoma (histopathologically). The study was conducted from September 2019 to February 2021.

MAIN OUTCOMES AND MEASURES

Identification and genotyping of a novel immunodeficiency-related RuV-associated granulomatous dermatitis.

RESULTS

Immunohistochemistry for RuV capsid protein and RT-PCR testing for RuV RNA revealed RuV in 4 discrete skin biopsies from different body sites. In addition, RuV RNA was detected in the patient's nasopharyngeal swabs by RT-PCR. The full viral genome was sequenced from the patient's skin biopsy (RVs/Philadelphia.PA.USA/46.19/GR, GenBank Accession #MT249313). The patient was ultimately diagnosed with a novel RuV-associated granulomatous dermatitis.

CONCLUSIONS AND RELEVANCE

The findings of this study suggest that clinicians and pathologists may consider RuV-associated granulomatous dermatitis during evaluation of a patient because it might have implications for the diagnosis of cutaneous sarcoidosis, with RuV serving as a potential antigenic trigger, and for the diagnosis of granulomatous cutaneous T-cell lymphoma, with histopathologic features that may prompt an evaluation for immunodeficiency and/or RuV.

摘要

重要性

免疫缺陷相关的疫苗衍生风疹病毒(RuV)作为一种抗原触发因素,导致皮肤和内脏肉芽肿,这是一种在接受免疫抑制剂治疗的儿童和年轻成年人中最近描述的罕见现象。

目的

进行全面的临床、组织学、免疫学、分子和基因组评估,以阐明一名成年患者非典型皮肤肉芽肿的潜在原因。

设计、地点和参与者:对提交给疾病控制与预防中心的皮肤活检、鼻咽拭子和血清样本进行了前瞻性评估,使用实时逆转录酶聚合酶链反应(RT-PCR)和病毒基因组测序来评估 RuV。这些样本来自一名 70 多岁的男性,他患有广泛的皮肤肉芽肿,既有皮肤结节病的临床表现(临床上),也有 CD8+肉芽肿性皮肤 T 细胞淋巴瘤的组织病理学表现。该研究于 2019 年 9 月至 2021 年 2 月进行。

主要结果和措施

鉴定和基因分型一种新的与免疫缺陷相关的 RuV 相关的肉芽肿性皮炎。

结果

对风疹病毒衣壳蛋白的免疫组织化学染色和对 RuV RNA 的 RT-PCR 检测显示,在来自不同身体部位的 4 个离散皮肤活检中均存在 RuV。此外,通过 RT-PCR 在患者的鼻咽拭子中检测到 RuV RNA。从患者的皮肤活检中对完整的病毒基因组进行测序(RVs/Philadelphia.PA.USA/46.19/GR,GenBank 登录号#MT249313)。该患者最终被诊断为一种新的与 RuV 相关的肉芽肿性皮炎。

结论和相关性

本研究结果表明,临床医生和病理学家在评估患者时可能会考虑到与 RuV 相关的肉芽肿性皮炎,因为它可能对皮肤结节病的诊断有影响,RuV 可能作为一种潜在的抗原触发因素,也可能对肉芽肿性皮肤 T 细胞淋巴瘤的诊断有影响,其组织病理学特征可能提示免疫缺陷和/或 RuV 的评估。

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