Epstein–Barr virus‐positive monoclonal lymphoplasmacytic proliferation associated with neurosyphilis in an immunocompetent patient: A case report
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 neurosyphili...
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creator | Hibiya, Takashi Nagahama, Kiyotaka Matsumoto, Yoshie Saito, Kuniaki Sasaki, Nobuyoshi Kobayashi, Keiichi Otsu, Akiyasu Shimasaki, Teppei Takeuchi, Kengo Shiokawa, Yoshiaki Nagane, Motoo Shibahara, Junji |
description | 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. |
doi_str_mv | 10.1111/neup.12934 |
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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.</description><identifier>ISSN: 0919-6544</identifier><identifier>EISSN: 1440-1789</identifier><identifier>DOI: 10.1111/neup.12934</identifier><identifier>PMID: 37424259</identifier><language>eng</language><publisher>Melbourne: John Wiley & Sons Australia, Ltd</publisher><subject>abscess ; Abscess - complications ; Abscesses ; Adult ; Antibiotics ; Cell Proliferation ; Central nervous system ; Cerebrum ; Epstein-Barr virus ; Epstein-Barr Virus Infections - complications ; Epstein–Barr virus infections ; Frontal lobe ; Herpesvirus 4, Human ; Humans ; Hybridization ; Immunocompetence ; Immunoproliferative diseases ; Infectious diseases ; Lesions ; Lymphocytes ; Lymphoid cells ; lymphoproliferative disorders ; Magnetic resonance imaging ; Male ; Meningitis ; Nervous system ; Neuroimaging ; Neurosyphilis ; Neurosyphilis - complications ; Neurosyphilis - diagnosis ; Patients ; Spirochetes ; Syphilis ; Syphilis - complications ; Treponema pallidum</subject><ispartof>Neuropathology, 2024-04, Vol.44 (2), p.104-108</ispartof><rights>2023 Japanese Society of Neuropathology.</rights><rights>2024 Japanese Society of Neuropathology</rights><lds50>peer_reviewed</lds50><woscitedreferencessubscribed>false</woscitedreferencessubscribed><cites>FETCH-LOGICAL-c2834-c3b9182bcd2365e7708ebfaa58a051c74cb0b7785c43cc6c3f194a2c4aaccc773</cites><orcidid>0000-0002-0018-1652 ; 0000-0001-6243-139X</orcidid></display><links><openurl>$$Topenurl_article</openurl><openurlfulltext>$$Topenurlfull_article</openurlfulltext><thumbnail>$$Tsyndetics_thumb_exl</thumbnail><linktopdf>$$Uhttps://onlinelibrary.wiley.com/doi/pdf/10.1111%2Fneup.12934$$EPDF$$P50$$Gwiley$$H</linktopdf><linktohtml>$$Uhttps://onlinelibrary.wiley.com/doi/full/10.1111%2Fneup.12934$$EHTML$$P50$$Gwiley$$H</linktohtml><link.rule.ids>314,780,784,1417,27924,27925,45574,45575</link.rule.ids><backlink>$$Uhttps://www.ncbi.nlm.nih.gov/pubmed/37424259$$D View this record in MEDLINE/PubMed$$Hfree_for_read</backlink></links><search><creatorcontrib>Hibiya, Takashi</creatorcontrib><creatorcontrib>Nagahama, Kiyotaka</creatorcontrib><creatorcontrib>Matsumoto, Yoshie</creatorcontrib><creatorcontrib>Saito, Kuniaki</creatorcontrib><creatorcontrib>Sasaki, Nobuyoshi</creatorcontrib><creatorcontrib>Kobayashi, Keiichi</creatorcontrib><creatorcontrib>Otsu, Akiyasu</creatorcontrib><creatorcontrib>Shimasaki, Teppei</creatorcontrib><creatorcontrib>Takeuchi, Kengo</creatorcontrib><creatorcontrib>Shiokawa, Yoshiaki</creatorcontrib><creatorcontrib>Nagane, Motoo</creatorcontrib><creatorcontrib>Shibahara, Junji</creatorcontrib><title>Epstein–Barr virus‐positive monoclonal lymphoplasmacytic proliferation associated with neurosyphilis in an immunocompetent patient: A case report</title><title>Neuropathology</title><addtitle>Neuropathology</addtitle><description>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.</description><subject>abscess</subject><subject>Abscess - complications</subject><subject>Abscesses</subject><subject>Adult</subject><subject>Antibiotics</subject><subject>Cell Proliferation</subject><subject>Central nervous system</subject><subject>Cerebrum</subject><subject>Epstein-Barr virus</subject><subject>Epstein-Barr Virus Infections - complications</subject><subject>Epstein–Barr virus infections</subject><subject>Frontal lobe</subject><subject>Herpesvirus 4, Human</subject><subject>Humans</subject><subject>Hybridization</subject><subject>Immunocompetence</subject><subject>Immunoproliferative diseases</subject><subject>Infectious diseases</subject><subject>Lesions</subject><subject>Lymphocytes</subject><subject>Lymphoid cells</subject><subject>lymphoproliferative disorders</subject><subject>Magnetic resonance imaging</subject><subject>Male</subject><subject>Meningitis</subject><subject>Nervous system</subject><subject>Neuroimaging</subject><subject>Neurosyphilis</subject><subject>Neurosyphilis - complications</subject><subject>Neurosyphilis - diagnosis</subject><subject>Patients</subject><subject>Spirochetes</subject><subject>Syphilis</subject><subject>Syphilis - complications</subject><subject>Treponema pallidum</subject><issn>0919-6544</issn><issn>1440-1789</issn><fulltext>true</fulltext><rsrctype>article</rsrctype><creationdate>2024</creationdate><recordtype>article</recordtype><sourceid>EIF</sourceid><recordid>eNp9kc1O3DAURq2Kqkyn3fQBKkvsKmXqv4xjdoCmgISARVlHzh1HY5TExnYGZccjVKr6gjwJpgMsuZu7OT66nz-EvlGyoHl-Dmb0C8oUFx_QjApBCiortYdmRFFVLEsh9tHnGG8JoVKx6hPa51IwwUo1Q_9WPiZjh8eHv8c6BLy1YYyPD3-8izbZrcG9Gxx0btAd7qbeb5zvdOw1TMkC9sF1tjVBJ-sGrGN0YHUya3xv0wbnu4KLk9_YzkZsMzBg2_djFrrem2SGhH1-mvchPsKgo8HBeBfSF_Sx1V00X1_2HN38Wv0-OSsurk7PT44uCmAVFwXwRtGKNbBmfFkaKUllmlbrstKkpCAFNKSRsipBcIAl8JYqoRkIrQFASj5HBztvDnI3mpjqWzeGnDXWnDCplpxVNFM_dhTkODGYtvbB9jpMNSX1cwP1cwP1_wYy_P1FOTa9Wb-hr1-eAboD7m1npndU9eXq5nonfQKU9Zht</recordid><startdate>202404</startdate><enddate>202404</enddate><creator>Hibiya, Takashi</creator><creator>Nagahama, Kiyotaka</creator><creator>Matsumoto, Yoshie</creator><creator>Saito, Kuniaki</creator><creator>Sasaki, Nobuyoshi</creator><creator>Kobayashi, Keiichi</creator><creator>Otsu, Akiyasu</creator><creator>Shimasaki, Teppei</creator><creator>Takeuchi, Kengo</creator><creator>Shiokawa, Yoshiaki</creator><creator>Nagane, Motoo</creator><creator>Shibahara, Junji</creator><general>John Wiley & Sons Australia, Ltd</general><general>Wiley Subscription Services, Inc</general><scope>CGR</scope><scope>CUY</scope><scope>CVF</scope><scope>ECM</scope><scope>EIF</scope><scope>NPM</scope><scope>AAYXX</scope><scope>CITATION</scope><scope>7TK</scope><scope>K9.</scope><orcidid>https://orcid.org/0000-0002-0018-1652</orcidid><orcidid>https://orcid.org/0000-0001-6243-139X</orcidid></search><sort><creationdate>202404</creationdate><title>Epstein–Barr virus‐positive monoclonal lymphoplasmacytic proliferation associated with neurosyphilis in an immunocompetent patient: A case report</title><author>Hibiya, Takashi ; Nagahama, Kiyotaka ; Matsumoto, Yoshie ; Saito, Kuniaki ; Sasaki, Nobuyoshi ; Kobayashi, Keiichi ; Otsu, Akiyasu ; Shimasaki, Teppei ; Takeuchi, Kengo ; Shiokawa, Yoshiaki ; Nagane, Motoo ; Shibahara, Junji</author></sort><facets><frbrtype>5</frbrtype><frbrgroupid>cdi_FETCH-LOGICAL-c2834-c3b9182bcd2365e7708ebfaa58a051c74cb0b7785c43cc6c3f194a2c4aaccc773</frbrgroupid><rsrctype>articles</rsrctype><prefilter>articles</prefilter><language>eng</language><creationdate>2024</creationdate><topic>abscess</topic><topic>Abscess - complications</topic><topic>Abscesses</topic><topic>Adult</topic><topic>Antibiotics</topic><topic>Cell Proliferation</topic><topic>Central nervous system</topic><topic>Cerebrum</topic><topic>Epstein-Barr virus</topic><topic>Epstein-Barr Virus Infections - complications</topic><topic>Epstein–Barr virus infections</topic><topic>Frontal lobe</topic><topic>Herpesvirus 4, Human</topic><topic>Humans</topic><topic>Hybridization</topic><topic>Immunocompetence</topic><topic>Immunoproliferative diseases</topic><topic>Infectious diseases</topic><topic>Lesions</topic><topic>Lymphocytes</topic><topic>Lymphoid cells</topic><topic>lymphoproliferative disorders</topic><topic>Magnetic resonance imaging</topic><topic>Male</topic><topic>Meningitis</topic><topic>Nervous system</topic><topic>Neuroimaging</topic><topic>Neurosyphilis</topic><topic>Neurosyphilis - complications</topic><topic>Neurosyphilis - diagnosis</topic><topic>Patients</topic><topic>Spirochetes</topic><topic>Syphilis</topic><topic>Syphilis - complications</topic><topic>Treponema pallidum</topic><toplevel>peer_reviewed</toplevel><toplevel>online_resources</toplevel><creatorcontrib>Hibiya, Takashi</creatorcontrib><creatorcontrib>Nagahama, Kiyotaka</creatorcontrib><creatorcontrib>Matsumoto, Yoshie</creatorcontrib><creatorcontrib>Saito, Kuniaki</creatorcontrib><creatorcontrib>Sasaki, Nobuyoshi</creatorcontrib><creatorcontrib>Kobayashi, Keiichi</creatorcontrib><creatorcontrib>Otsu, Akiyasu</creatorcontrib><creatorcontrib>Shimasaki, Teppei</creatorcontrib><creatorcontrib>Takeuchi, Kengo</creatorcontrib><creatorcontrib>Shiokawa, Yoshiaki</creatorcontrib><creatorcontrib>Nagane, Motoo</creatorcontrib><creatorcontrib>Shibahara, Junji</creatorcontrib><collection>Medline</collection><collection>MEDLINE</collection><collection>MEDLINE (Ovid)</collection><collection>MEDLINE</collection><collection>MEDLINE</collection><collection>PubMed</collection><collection>CrossRef</collection><collection>Neurosciences Abstracts</collection><collection>ProQuest Health & Medical Complete (Alumni)</collection><jtitle>Neuropathology</jtitle></facets><delivery><delcategory>Remote Search Resource</delcategory><fulltext>fulltext</fulltext></delivery><addata><au>Hibiya, Takashi</au><au>Nagahama, Kiyotaka</au><au>Matsumoto, Yoshie</au><au>Saito, Kuniaki</au><au>Sasaki, Nobuyoshi</au><au>Kobayashi, Keiichi</au><au>Otsu, Akiyasu</au><au>Shimasaki, Teppei</au><au>Takeuchi, Kengo</au><au>Shiokawa, Yoshiaki</au><au>Nagane, Motoo</au><au>Shibahara, Junji</au><format>journal</format><genre>article</genre><ristype>JOUR</ristype><atitle>Epstein–Barr virus‐positive monoclonal lymphoplasmacytic proliferation associated with neurosyphilis in an immunocompetent patient: A case report</atitle><jtitle>Neuropathology</jtitle><addtitle>Neuropathology</addtitle><date>2024-04</date><risdate>2024</risdate><volume>44</volume><issue>2</issue><spage>104</spage><epage>108</epage><pages>104-108</pages><issn>0919-6544</issn><eissn>1440-1789</eissn><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.</abstract><cop>Melbourne</cop><pub>John Wiley & Sons Australia, Ltd</pub><pmid>37424259</pmid><doi>10.1111/neup.12934</doi><tpages>5</tpages><orcidid>https://orcid.org/0000-0002-0018-1652</orcidid><orcidid>https://orcid.org/0000-0001-6243-139X</orcidid></addata></record> |
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subjects | abscess Abscess - complications Abscesses Adult Antibiotics Cell Proliferation Central nervous system Cerebrum Epstein-Barr virus Epstein-Barr Virus Infections - complications Epstein–Barr virus infections Frontal lobe Herpesvirus 4, Human Humans Hybridization Immunocompetence Immunoproliferative diseases Infectious diseases Lesions Lymphocytes Lymphoid cells lymphoproliferative disorders Magnetic resonance imaging Male Meningitis Nervous system Neuroimaging Neurosyphilis Neurosyphilis - complications Neurosyphilis - diagnosis Patients Spirochetes Syphilis Syphilis - complications Treponema pallidum |
title | Epstein–Barr virus‐positive monoclonal lymphoplasmacytic proliferation associated with neurosyphilis in an immunocompetent patient: A case report |
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