Epstein-Barr virus-positive monoclonal lymphoplasmacytic proliferation associated with neurosyphilis in an immunocompetent patient: A case report.

IF 1.3 4区 医学 Q4 CLINICAL NEUROLOGY
Neuropathology Pub Date : 2024-04-01 Epub Date: 2023-07-09 DOI:10.1111/neup.12934
Takashi Hibiya, Kiyotaka Nagahama, Yoshie Matsumoto, Kuniaki Saito, Nobuyoshi Sasaki, Keiichi Kobayashi, Akiyasu Otsu, Teppei Shimasaki, Kengo Takeuchi, Yoshiaki Shiokawa, Motoo Nagane, Junji Shibahara
{"title":"Epstein-Barr virus-positive monoclonal lymphoplasmacytic proliferation associated with neurosyphilis in an immunocompetent patient: A case report.","authors":"Takashi Hibiya, Kiyotaka Nagahama, Yoshie Matsumoto, Kuniaki Saito, Nobuyoshi Sasaki, Keiichi Kobayashi, Akiyasu Otsu, Teppei Shimasaki, Kengo Takeuchi, Yoshiaki Shiokawa, Motoo Nagane, Junji Shibahara","doi":"10.1111/neup.12934","DOIUrl":null,"url":null,"abstract":"<p><p>Syphilis is an infectious disease caused by the spirochete bacterium Treponema pallidum. Neurosyphilis results from the infection of the nervous system with Treponema pallidum, which can occur at any stage of syphilis. Neurosyphilis is often overlooked because of its rarity. Early-stage neurosyphilis with brain mass formation is rare. We present a case of early-stage neurosyphilis with prominent Epstein-Barr virus (EBV)-positive monoclonal lymphoplasmacytic proliferation in an immunocompetent patient. A 36-year-old man presented with a chief complaint of a progressively worsening headache, a newly developed skin rash, and a fever. Magnetic resonance imaging showed a mass lesion, which measured 18 mm in diameter, in the left frontal lobe of the cerebrum. The patient underwent an emergency operation to remove the abscess. A pathological investigation revealed complex findings. There was an abscess in the cerebrum. Lymphoplasmacytic meningitis was also noted. In addition, a vaguely nodular lesion, which was composed of plasmacytoid and lymphoid cells, was observed around the abscess. Immunohistochemically, an anti-Treponema pallidum antibody revealed numerous Treponemas around the abscess. In situ hybridization revealed that the plasmacytoid and lymphoid cells were Epstein-Barr encoding region (EBER)-positive; κ-positive cells were significantly more prevalent than λ-positive cells, suggesting light-chain restriction. Postoperatively, parenteral antibiotics were administered for four weeks. The patient has been free of recurrence for two years since the surgery. No association between neurosyphilis and EBV-positive lymphoplasmacytic proliferation has ever been reported. Mass formation in early-stage neurosyphilis is an exceptionally rare event. The present case indicates that in syphilis patients, lymphoproliferative disorders that lead to mass formation may be caused by concomitant EBV reactivation. Furthermore, when treating patients with mass lesions of the central nervous system, it is important to check their medical history and perform laboratory screening for infectious diseases to avoid overlooking syphilis infections.</p>","PeriodicalId":19204,"journal":{"name":"Neuropathology","volume":" ","pages":"104-108"},"PeriodicalIF":1.3000,"publicationDate":"2024-04-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":"0","resultStr":null,"platform":"Semanticscholar","paperid":null,"PeriodicalName":"Neuropathology","FirstCategoryId":"3","ListUrlMain":"https://doi.org/10.1111/neup.12934","RegionNum":4,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":null,"EPubDate":"2023/7/9 0:00:00","PubModel":"Epub","JCR":"Q4","JCRName":"CLINICAL NEUROLOGY","Score":null,"Total":0}
引用次数: 0

Abstract

Syphilis is an infectious disease caused by the spirochete bacterium Treponema pallidum. Neurosyphilis results from the infection of the nervous system with Treponema pallidum, which can occur at any stage of syphilis. Neurosyphilis is often overlooked because of its rarity. Early-stage neurosyphilis with brain mass formation is rare. We present a case of early-stage neurosyphilis with prominent Epstein-Barr virus (EBV)-positive monoclonal lymphoplasmacytic proliferation in an immunocompetent patient. A 36-year-old man presented with a chief complaint of a progressively worsening headache, a newly developed skin rash, and a fever. Magnetic resonance imaging showed a mass lesion, which measured 18 mm in diameter, in the left frontal lobe of the cerebrum. The patient underwent an emergency operation to remove the abscess. A pathological investigation revealed complex findings. There was an abscess in the cerebrum. Lymphoplasmacytic meningitis was also noted. In addition, a vaguely nodular lesion, which was composed of plasmacytoid and lymphoid cells, was observed around the abscess. Immunohistochemically, an anti-Treponema pallidum antibody revealed numerous Treponemas around the abscess. In situ hybridization revealed that the plasmacytoid and lymphoid cells were Epstein-Barr encoding region (EBER)-positive; κ-positive cells were significantly more prevalent than λ-positive cells, suggesting light-chain restriction. Postoperatively, parenteral antibiotics were administered for four weeks. The patient has been free of recurrence for two years since the surgery. No association between neurosyphilis and EBV-positive lymphoplasmacytic proliferation has ever been reported. Mass formation in early-stage neurosyphilis is an exceptionally rare event. The present case indicates that in syphilis patients, lymphoproliferative disorders that lead to mass formation may be caused by concomitant EBV reactivation. Furthermore, when treating patients with mass lesions of the central nervous system, it is important to check their medical history and perform laboratory screening for infectious diseases to avoid overlooking syphilis infections.

一名免疫功能正常的患者出现了与神经梅毒相关的 Epstein-Barr 病毒阳性单克隆淋巴浆细胞增生:病例报告。
梅毒是一种由苍白螺旋体引起的传染性疾病。神经梅毒是苍白螺旋体感染神经系统的结果,可发生在梅毒的任何阶段。神经梅毒因其罕见而常常被忽视。早期神经梅毒伴有脑肿块形成的病例十分罕见。我们报告了一例免疫功能正常的早期神经梅毒患者,其脑部有明显的爱泼斯坦-巴氏病毒(EBV)阳性单克隆淋巴浆细胞增生。一名 36 岁的男子主诉头痛逐渐加重、新发皮疹和发热。磁共振成像显示,左侧大脑额叶有一个直径为18毫米的肿块病变。患者接受了切除脓肿的紧急手术。病理检查显示了复杂的结果。大脑中有一个脓肿。还发现了淋巴浆细胞性脑膜炎。此外,在脓肿周围还发现了由浆细胞和淋巴细胞组成的模糊结节性病变。免疫组化检查发现,脓肿周围有大量的苍白盘尾丝虫抗体。原位杂交显示,浆细胞和淋巴细胞呈爱泼斯坦-巴氏菌编码区(EBER)阳性;κ阳性细胞明显多于λ阳性细胞,表明轻链受限。术后,患者接受了为期四周的肠外抗生素治疗。患者术后两年未再复发。神经梅毒与EB病毒阳性淋巴浆细胞增生之间的关系尚未见报道。早期神经梅毒形成肿块的情况非常罕见。本病例表明,在梅毒患者中,导致肿块形成的淋巴组织增生性疾病可能是由同时存在的EB病毒再激活引起的。此外,在治疗中枢神经系统肿块病变的患者时,必须检查其病史并进行传染病实验室筛查,以避免忽视梅毒感染。
本文章由计算机程序翻译,如有差异,请以英文原文为准。
求助全文
约1分钟内获得全文 求助全文
来源期刊
Neuropathology
Neuropathology 医学-病理学
CiteScore
4.10
自引率
4.30%
发文量
105
审稿时长
6-12 weeks
期刊介绍: Neuropathology is an international journal sponsored by the Japanese Society of Neuropathology and publishes peer-reviewed original papers dealing with all aspects of human and experimental neuropathology and related fields of research. The Journal aims to promote the international exchange of results and encourages authors from all countries to submit papers in the following categories: Original Articles, Case Reports, Short Communications, Occasional Reviews, Editorials and Letters to the Editor. All articles are peer-reviewed by at least two researchers expert in the field of the submitted paper.
×
引用
GB/T 7714-2015
复制
MLA
复制
APA
复制
导出至
BibTeX EndNote RefMan NoteFirst NoteExpress
×
提示
您的信息不完整,为了账户安全,请先补充。
现在去补充
×
提示
您因"违规操作"
具体请查看互助需知
我知道了
×
提示
确定
请完成安全验证×
copy
已复制链接
快去分享给好友吧!
我知道了
右上角分享
点击右上角分享
0
联系我们:info@booksci.cn Book学术提供免费学术资源搜索服务,方便国内外学者检索中英文文献。致力于提供最便捷和优质的服务体验。 Copyright © 2023 布克学术 All rights reserved.
京ICP备2023020795号-1
ghs 京公网安备 11010802042870号
Book学术文献互助
Book学术文献互助群
群 号:481959085
Book学术官方微信