Isolated EBV lymphoproliferative disease in a child with Wiskott-Aldrich syndrome manifesting as cutaneous lymphomatoid granulomatosis and responsive to anti-CD20 immunotherapy
Patients with primary immunodeficiencies such as the Wiskott-Aldrich syndrome (WAS) are prone to develop Epstein-Barr virus (EBV) related lymphoproliferative disorders (LPDs). EBV LPD is most frequently seen in patients receiving immunosuppressive treatment after organ transplantation (post-transpla...
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| description | Patients with primary immunodeficiencies such as the Wiskott-Aldrich syndrome (WAS) are prone to develop Epstein-Barr virus (EBV) related lymphoproliferative disorders (LPDs). EBV LPD is most frequently seen in patients receiving immunosuppressive treatment after organ transplantation (post-transplant lymphoproliferative disorder), but can also arise in the primary immunodeficiencies. Typically, EBV LPD presents as a diffuse systemic disease with lymphadenopathy and organ involvement. A rare angiocentric and angiodestructive form of EBV associated B cell LPD, lymphomatoid granulomatosis (LyG), has also been reported in association with WAS. LyG most commonly involves the lung, but can also be seen in brain, kidney, liver, and skin. This report describes the case of a 16 year old boy with WAS who presented with an isolated non-healing ulcerating skin lesion. Biopsy revealed an EBV related LPD with the histological features of LyG. This cutaneous lesion responded dramatically to treatment with specific anti-CD20 immunotherapy and the patient remains clinically free of LPD at 18 months. |
| doi_str_mv | 10.1136/jcp.56.7.555 |
| format | Article |
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EBV LPD is most frequently seen in patients receiving immunosuppressive treatment after organ transplantation (post-transplant lymphoproliferative disorder), but can also arise in the primary immunodeficiencies. Typically, EBV LPD presents as a diffuse systemic disease with lymphadenopathy and organ involvement. A rare angiocentric and angiodestructive form of EBV associated B cell LPD, lymphomatoid granulomatosis (LyG), has also been reported in association with WAS. LyG most commonly involves the lung, but can also be seen in brain, kidney, liver, and skin. This report describes the case of a 16 year old boy with WAS who presented with an isolated non-healing ulcerating skin lesion. Biopsy revealed an EBV related LPD with the histological features of LyG. This cutaneous lesion responded dramatically to treatment with specific anti-CD20 immunotherapy and the patient remains clinically free of LPD at 18 months.</description><identifier>ISSN: 0021-9746</identifier><identifier>EISSN: 1472-4146</identifier><identifier>DOI: 10.1136/jcp.56.7.555</identifier><identifier>PMID: 12835306</identifier><identifier>CODEN: JCPAAK</identifier><language>eng</language><publisher>London: BMJ Publishing Group Ltd and Association of Clinical Pathologists</publisher><subject>Adolescent ; anti-CD20 ; Antibodies, Monoclonal - therapeutic use ; Antibodies, Monoclonal, Murine-Derived ; Antigens, CD20 - immunology ; Biological and medical sciences ; Bone marrow ; Care and treatment ; Case Reports ; Causes of ; Cytotoxicity ; Development and progression ; EBER ; EBV ; Epstein-Barr encoded viral RNAs ; Epstein-Barr virus ; Epstein-Barr Virus Infections - complications ; Epstein-Barr Virus Infections - pathology ; Epstein-Barr Virus Infections - therapy ; Genes ; Hematologic and hematopoietic diseases ; Histology ; Humans ; Immunoglobulins ; Immunotherapy ; in situ hybridisation ; Infections ; ISH ; Leukemias. Malignant lymphomas. Malignant reticulosis. Myelofibrosis ; LPD ; LyG ; Lymphocytes ; Lymphoma ; lymphomatoid granulomatosis ; Lymphomatoid Granulomatosis - etiology ; Lymphomatoid Granulomatosis - pathology ; Lymphomatoid Granulomatosis - therapy ; lymphoproliferative disorder ; Lymphoproliferative disorders ; Lymphoproliferative Disorders - pathology ; Lymphoproliferative Disorders - therapy ; Lymphoproliferative Disorders - virology ; Male ; Medical sciences ; Platelet diseases and coagulopathies ; post-transplant lymphoproliferative disorder ; PTLD ; Risk factors ; Rituximab ; Skin Diseases - etiology ; Skin Diseases - pathology ; Skin Diseases - therapy ; Transplants & implants ; WAS ; Wiskott-Aldrich ; Wiskott-Aldrich syndrome ; Wiskott-Aldrich Syndrome - pathology ; Wiskott-Aldrich Syndrome - therapy ; Wiskott-Aldrich Syndrome - virology</subject><ispartof>Journal of clinical pathology, 2003-07, Vol.56 (7), p.555-557</ispartof><rights>Copyright 2003 Journal of Clinical Pathology</rights><rights>2003 INIST-CNRS</rights><rights>COPYRIGHT 2003 BMJ Publishing Group Ltd.</rights><rights>Copyright: 2003 Copyright 2003 Journal of Clinical Pathology</rights><rights>Copyright © Copyright 2003 Journal of Clinical Pathology 2003</rights><lds50>peer_reviewed</lds50><oa>free_for_read</oa><woscitedreferencessubscribed>false</woscitedreferencessubscribed><citedby>FETCH-LOGICAL-b585t-c2fdf1d3488d1f50389d05659be550db75f44bd644b454fd54a2fe640f09b3803</citedby></display><links><openurl>$$Topenurl_article</openurl><openurlfulltext>$$Topenurlfull_article</openurlfulltext><thumbnail>$$Tsyndetics_thumb_exl</thumbnail><linktopdf>$$Uhttps://www.ncbi.nlm.nih.gov/pmc/articles/PMC1769998/pdf/$$EPDF$$P50$$Gpubmedcentral$$H</linktopdf><linktohtml>$$Uhttps://www.ncbi.nlm.nih.gov/pmc/articles/PMC1769998/$$EHTML$$P50$$Gpubmedcentral$$H</linktohtml><link.rule.ids>230,314,723,776,780,881,27901,27902,53766,53768</link.rule.ids><backlink>$$Uhttp://pascal-francis.inist.fr/vibad/index.php?action=getRecordDetail&idt=14912623$$DView record in Pascal Francis$$Hfree_for_read</backlink><backlink>$$Uhttps://www.ncbi.nlm.nih.gov/pubmed/12835306$$D View this record in MEDLINE/PubMed$$Hfree_for_read</backlink></links><search><creatorcontrib>Sebire, N J</creatorcontrib><creatorcontrib>Haselden, S</creatorcontrib><creatorcontrib>Malone, M</creatorcontrib><creatorcontrib>Davies, E G</creatorcontrib><creatorcontrib>Ramsay, A D</creatorcontrib><title>Isolated EBV lymphoproliferative disease in a child with Wiskott-Aldrich syndrome manifesting as cutaneous lymphomatoid granulomatosis and responsive to anti-CD20 immunotherapy</title><title>Journal of clinical pathology</title><addtitle>J Clin Pathol</addtitle><description>Patients with primary immunodeficiencies such as the Wiskott-Aldrich syndrome (WAS) are prone to develop Epstein-Barr virus (EBV) related lymphoproliferative disorders (LPDs). EBV LPD is most frequently seen in patients receiving immunosuppressive treatment after organ transplantation (post-transplant lymphoproliferative disorder), but can also arise in the primary immunodeficiencies. Typically, EBV LPD presents as a diffuse systemic disease with lymphadenopathy and organ involvement. A rare angiocentric and angiodestructive form of EBV associated B cell LPD, lymphomatoid granulomatosis (LyG), has also been reported in association with WAS. LyG most commonly involves the lung, but can also be seen in brain, kidney, liver, and skin. This report describes the case of a 16 year old boy with WAS who presented with an isolated non-healing ulcerating skin lesion. Biopsy revealed an EBV related LPD with the histological features of LyG. This cutaneous lesion responded dramatically to treatment with specific anti-CD20 immunotherapy and the patient remains clinically free of LPD at 18 months.</description><subject>Adolescent</subject><subject>anti-CD20</subject><subject>Antibodies, Monoclonal - therapeutic use</subject><subject>Antibodies, Monoclonal, Murine-Derived</subject><subject>Antigens, CD20 - immunology</subject><subject>Biological and medical sciences</subject><subject>Bone marrow</subject><subject>Care and treatment</subject><subject>Case Reports</subject><subject>Causes of</subject><subject>Cytotoxicity</subject><subject>Development and progression</subject><subject>EBER</subject><subject>EBV</subject><subject>Epstein-Barr encoded viral RNAs</subject><subject>Epstein-Barr virus</subject><subject>Epstein-Barr Virus Infections - complications</subject><subject>Epstein-Barr Virus Infections - pathology</subject><subject>Epstein-Barr Virus Infections - therapy</subject><subject>Genes</subject><subject>Hematologic and hematopoietic diseases</subject><subject>Histology</subject><subject>Humans</subject><subject>Immunoglobulins</subject><subject>Immunotherapy</subject><subject>in situ hybridisation</subject><subject>Infections</subject><subject>ISH</subject><subject>Leukemias. Malignant lymphomas. Malignant reticulosis. Myelofibrosis</subject><subject>LPD</subject><subject>LyG</subject><subject>Lymphocytes</subject><subject>Lymphoma</subject><subject>lymphomatoid granulomatosis</subject><subject>Lymphomatoid Granulomatosis - etiology</subject><subject>Lymphomatoid Granulomatosis - pathology</subject><subject>Lymphomatoid Granulomatosis - therapy</subject><subject>lymphoproliferative disorder</subject><subject>Lymphoproliferative disorders</subject><subject>Lymphoproliferative Disorders - pathology</subject><subject>Lymphoproliferative Disorders - therapy</subject><subject>Lymphoproliferative Disorders - virology</subject><subject>Male</subject><subject>Medical sciences</subject><subject>Platelet diseases and coagulopathies</subject><subject>post-transplant lymphoproliferative disorder</subject><subject>PTLD</subject><subject>Risk factors</subject><subject>Rituximab</subject><subject>Skin Diseases - etiology</subject><subject>Skin Diseases - pathology</subject><subject>Skin Diseases - therapy</subject><subject>Transplants & implants</subject><subject>WAS</subject><subject>Wiskott-Aldrich</subject><subject>Wiskott-Aldrich syndrome</subject><subject>Wiskott-Aldrich Syndrome - pathology</subject><subject>Wiskott-Aldrich Syndrome - therapy</subject><subject>Wiskott-Aldrich Syndrome - virology</subject><issn>0021-9746</issn><issn>1472-4146</issn><fulltext>true</fulltext><rsrctype>article</rsrctype><creationdate>2003</creationdate><recordtype>article</recordtype><sourceid>EIF</sourceid><sourceid>BENPR</sourceid><recordid>eNp9kl1vFCEUhidGY9fqndeGxKg3zgozfMzcmNS1tU2q3uj2kjAD7LJlYASmuv_Knyh1N62axpBAODznPR-coniK4Byhmr7Z9OOc0DmbE0LuFTOEWVVihOn9YgZhhcqWYXpQPIpxAyGqGaofFgeoampSQzorfp5Fb0VSEhy_WwK7Hca1H4O3RqsgkrlSQJqoRFTAOCBAvzZWgu8mrcGFiZc-pfLIymD6NYhbJ4MfFBiEy94xGbcCIoJ-SsIpP8W9-iCSNxKsgnCT_X2LJgLhJAgqjt7F66DJZ0sy5eJ9BYEZhsn5tM4ZjdvHxQMtbFRP9udh8fXk-MvitDz__OFscXRedqQhqewrLTWSNW4aiTSBddNKSChpO0UIlB0jGuNO0rxhgrUkWFRaUQw1bLu6gfVh8XanO07doGSvXArC8jGYQYQt98Lwv1-cWfOVv-KI0bZtmyzwci8Q_Lcp94MPJvbK2l03OKtxxSimGXz-D7jxU3C5uKzVIFTlb0OZKnfUSljFjdM-R-1XyuWuWO-UNtl8hCBhlEGGMz-_g89LqsH0dzq83jn0wccYlL6pFUF-PWk8TxonlDOeJy3jz_7szy28H60MvNgDIvbC6vzfvYm3HG5RRav6tjATk_px8y7CJaesZoR_Wi54e_FxAU_gKV9m_tWO74bN_1P8BcYY_Aw</recordid><startdate>20030701</startdate><enddate>20030701</enddate><creator>Sebire, N J</creator><creator>Haselden, S</creator><creator>Malone, M</creator><creator>Davies, E G</creator><creator>Ramsay, A D</creator><general>BMJ Publishing Group Ltd and Association of Clinical Pathologists</general><general>BMJ</general><general>BMJ Publishing Group Ltd</general><general>BMJ Publishing Group LTD</general><general>Copyright 2003 Journal of Clinical Pathology</general><scope>BSCLL</scope><scope>IQODW</scope><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>3V.</scope><scope>7X7</scope><scope>7XB</scope><scope>88E</scope><scope>88I</scope><scope>8AF</scope><scope>8FI</scope><scope>8FJ</scope><scope>8FK</scope><scope>ABUWG</scope><scope>AFKRA</scope><scope>AZQEC</scope><scope>BENPR</scope><scope>BTHHO</scope><scope>CCPQU</scope><scope>DWQXO</scope><scope>FYUFA</scope><scope>GHDGH</scope><scope>GNUQQ</scope><scope>HCIFZ</scope><scope>K9.</scope><scope>M0S</scope><scope>M1P</scope><scope>M2P</scope><scope>PQEST</scope><scope>PQQKQ</scope><scope>PQUKI</scope><scope>PRINS</scope><scope>Q9U</scope><scope>7X8</scope><scope>5PM</scope></search><sort><creationdate>20030701</creationdate><title>Isolated EBV lymphoproliferative disease in a child with Wiskott-Aldrich syndrome manifesting as cutaneous lymphomatoid granulomatosis and responsive to anti-CD20 immunotherapy</title><author>Sebire, N J ; Haselden, S ; Malone, M ; Davies, E G ; Ramsay, A D</author></sort><facets><frbrtype>5</frbrtype><frbrgroupid>cdi_FETCH-LOGICAL-b585t-c2fdf1d3488d1f50389d05659be550db75f44bd644b454fd54a2fe640f09b3803</frbrgroupid><rsrctype>articles</rsrctype><prefilter>articles</prefilter><language>eng</language><creationdate>2003</creationdate><topic>Adolescent</topic><topic>anti-CD20</topic><topic>Antibodies, Monoclonal - therapeutic use</topic><topic>Antibodies, Monoclonal, Murine-Derived</topic><topic>Antigens, CD20 - immunology</topic><topic>Biological and medical sciences</topic><topic>Bone marrow</topic><topic>Care and treatment</topic><topic>Case Reports</topic><topic>Causes of</topic><topic>Cytotoxicity</topic><topic>Development and progression</topic><topic>EBER</topic><topic>EBV</topic><topic>Epstein-Barr encoded viral RNAs</topic><topic>Epstein-Barr virus</topic><topic>Epstein-Barr Virus Infections - complications</topic><topic>Epstein-Barr Virus Infections - pathology</topic><topic>Epstein-Barr Virus Infections - therapy</topic><topic>Genes</topic><topic>Hematologic and hematopoietic diseases</topic><topic>Histology</topic><topic>Humans</topic><topic>Immunoglobulins</topic><topic>Immunotherapy</topic><topic>in situ hybridisation</topic><topic>Infections</topic><topic>ISH</topic><topic>Leukemias. Malignant lymphomas. Malignant reticulosis. Myelofibrosis</topic><topic>LPD</topic><topic>LyG</topic><topic>Lymphocytes</topic><topic>Lymphoma</topic><topic>lymphomatoid granulomatosis</topic><topic>Lymphomatoid Granulomatosis - etiology</topic><topic>Lymphomatoid Granulomatosis - pathology</topic><topic>Lymphomatoid Granulomatosis - therapy</topic><topic>lymphoproliferative disorder</topic><topic>Lymphoproliferative disorders</topic><topic>Lymphoproliferative Disorders - pathology</topic><topic>Lymphoproliferative Disorders - therapy</topic><topic>Lymphoproliferative Disorders - virology</topic><topic>Male</topic><topic>Medical sciences</topic><topic>Platelet diseases and coagulopathies</topic><topic>post-transplant lymphoproliferative disorder</topic><topic>PTLD</topic><topic>Risk factors</topic><topic>Rituximab</topic><topic>Skin Diseases - etiology</topic><topic>Skin Diseases - pathology</topic><topic>Skin Diseases - therapy</topic><topic>Transplants & implants</topic><topic>WAS</topic><topic>Wiskott-Aldrich</topic><topic>Wiskott-Aldrich syndrome</topic><topic>Wiskott-Aldrich Syndrome - pathology</topic><topic>Wiskott-Aldrich Syndrome - therapy</topic><topic>Wiskott-Aldrich Syndrome - virology</topic><toplevel>peer_reviewed</toplevel><toplevel>online_resources</toplevel><creatorcontrib>Sebire, N J</creatorcontrib><creatorcontrib>Haselden, S</creatorcontrib><creatorcontrib>Malone, M</creatorcontrib><creatorcontrib>Davies, E G</creatorcontrib><creatorcontrib>Ramsay, A D</creatorcontrib><collection>Istex</collection><collection>Pascal-Francis</collection><collection>Medline</collection><collection>MEDLINE</collection><collection>MEDLINE (Ovid)</collection><collection>MEDLINE</collection><collection>MEDLINE</collection><collection>PubMed</collection><collection>CrossRef</collection><collection>ProQuest Central (Corporate)</collection><collection>Health & Medical Collection</collection><collection>ProQuest Central (purchase pre-March 2016)</collection><collection>Medical Database (Alumni Edition)</collection><collection>Science Database (Alumni Edition)</collection><collection>STEM Database</collection><collection>Hospital Premium Collection</collection><collection>Hospital Premium Collection (Alumni Edition)</collection><collection>ProQuest Central (Alumni) (purchase pre-March 2016)</collection><collection>ProQuest Central (Alumni Edition)</collection><collection>ProQuest Central UK/Ireland</collection><collection>ProQuest Central Essentials</collection><collection>ProQuest Central</collection><collection>BMJ Journals</collection><collection>ProQuest One Community College</collection><collection>ProQuest Central Korea</collection><collection>Health Research Premium Collection</collection><collection>Health Research Premium Collection (Alumni)</collection><collection>ProQuest Central Student</collection><collection>SciTech Premium Collection</collection><collection>ProQuest Health & Medical Complete (Alumni)</collection><collection>Health & Medical Collection (Alumni Edition)</collection><collection>Medical Database</collection><collection>Science Database</collection><collection>ProQuest One Academic Eastern Edition (DO NOT USE)</collection><collection>ProQuest One Academic</collection><collection>ProQuest One Academic UKI Edition</collection><collection>ProQuest Central China</collection><collection>ProQuest Central Basic</collection><collection>MEDLINE - Academic</collection><collection>PubMed Central (Full Participant titles)</collection><jtitle>Journal of clinical pathology</jtitle></facets><delivery><delcategory>Remote Search Resource</delcategory><fulltext>fulltext</fulltext></delivery><addata><au>Sebire, N J</au><au>Haselden, S</au><au>Malone, M</au><au>Davies, E G</au><au>Ramsay, A D</au><format>journal</format><genre>article</genre><ristype>JOUR</ristype><atitle>Isolated EBV lymphoproliferative disease in a child with Wiskott-Aldrich syndrome manifesting as cutaneous lymphomatoid granulomatosis and responsive to anti-CD20 immunotherapy</atitle><jtitle>Journal of clinical pathology</jtitle><addtitle>J Clin Pathol</addtitle><date>2003-07-01</date><risdate>2003</risdate><volume>56</volume><issue>7</issue><spage>555</spage><epage>557</epage><pages>555-557</pages><issn>0021-9746</issn><eissn>1472-4146</eissn><coden>JCPAAK</coden><abstract>Patients with primary immunodeficiencies such as the Wiskott-Aldrich syndrome (WAS) are prone to develop Epstein-Barr virus (EBV) related lymphoproliferative disorders (LPDs). EBV LPD is most frequently seen in patients receiving immunosuppressive treatment after organ transplantation (post-transplant lymphoproliferative disorder), but can also arise in the primary immunodeficiencies. Typically, EBV LPD presents as a diffuse systemic disease with lymphadenopathy and organ involvement. A rare angiocentric and angiodestructive form of EBV associated B cell LPD, lymphomatoid granulomatosis (LyG), has also been reported in association with WAS. LyG most commonly involves the lung, but can also be seen in brain, kidney, liver, and skin. This report describes the case of a 16 year old boy with WAS who presented with an isolated non-healing ulcerating skin lesion. Biopsy revealed an EBV related LPD with the histological features of LyG. This cutaneous lesion responded dramatically to treatment with specific anti-CD20 immunotherapy and the patient remains clinically free of LPD at 18 months.</abstract><cop>London</cop><pub>BMJ Publishing Group Ltd and Association of Clinical Pathologists</pub><pmid>12835306</pmid><doi>10.1136/jcp.56.7.555</doi><tpages>3</tpages><oa>free_for_read</oa></addata></record> |
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| subjects | Adolescent anti-CD20 Antibodies, Monoclonal - therapeutic use Antibodies, Monoclonal, Murine-Derived Antigens, CD20 - immunology Biological and medical sciences Bone marrow Care and treatment Case Reports Causes of Cytotoxicity Development and progression EBER EBV Epstein-Barr encoded viral RNAs Epstein-Barr virus Epstein-Barr Virus Infections - complications Epstein-Barr Virus Infections - pathology Epstein-Barr Virus Infections - therapy Genes Hematologic and hematopoietic diseases Histology Humans Immunoglobulins Immunotherapy in situ hybridisation Infections ISH Leukemias. Malignant lymphomas. Malignant reticulosis. Myelofibrosis LPD LyG Lymphocytes Lymphoma lymphomatoid granulomatosis Lymphomatoid Granulomatosis - etiology Lymphomatoid Granulomatosis - pathology Lymphomatoid Granulomatosis - therapy lymphoproliferative disorder Lymphoproliferative disorders Lymphoproliferative Disorders - pathology Lymphoproliferative Disorders - therapy Lymphoproliferative Disorders - virology Male Medical sciences Platelet diseases and coagulopathies post-transplant lymphoproliferative disorder PTLD Risk factors Rituximab Skin Diseases - etiology Skin Diseases - pathology Skin Diseases - therapy Transplants & implants WAS Wiskott-Aldrich Wiskott-Aldrich syndrome Wiskott-Aldrich Syndrome - pathology Wiskott-Aldrich Syndrome - therapy Wiskott-Aldrich Syndrome - virology |
| title | Isolated EBV lymphoproliferative disease in a child with Wiskott-Aldrich syndrome manifesting as cutaneous lymphomatoid granulomatosis and responsive to anti-CD20 immunotherapy |
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