Posttransplant lymphoproliferative disorder after umbilical cord blood transplantation in children
Reported are 7 cases of posttransplant lymphoproliferative disorder (PTLD) arising in children who received umbilical cord blood transplantation (UCBT). There were 4 females and 3 males with a median age of 3 years (range, 1-16 years). All 7 patients received UCBT, including 1 patient who received m...
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Veröffentlicht in: | The American journal of surgical pathology 2006-03, Vol.30 (3), p.328-336 |
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creator | GONG, Jerald Z BAYERL, Michael G BUCKLEY, Patrick J SANDHAUS, Linda M SEBASTIAN, Siby REHDER, Catherine W ROUTBORT, Mark LAGOO, Anand S SZABOLCS, Paul CHIU, Jeanie COMITO, Melanie |
description | Reported are 7 cases of posttransplant lymphoproliferative disorder (PTLD) arising in children who received umbilical cord blood transplantation (UCBT). There were 4 females and 3 males with a median age of 3 years (range, 1-16 years). All 7 patients received UCBT, including 1 patient who received multiple units and 1 transplanted under nonmyeloablative condition. The time interval from UCBT to PTLD averaged 4 months (range, 2 weeks to 9 months). Patients typically presented with high-stage disease with visceral organ involvement. Histology of the PTLDs showed monomorphic morphology in 5 cases and polymorphic morphology in the remaining 2 cases. Bone marrow biopsies were performed in 3 cases and were negative for PTLD. Epstein-Barr virus (EBV) was detected in the PTLD in all 7 patients by in situ hybridization. Evidence of past EBV infection was found in the recipients, but the EBV genome was not detected in the donor cord blood samples, suggesting that donor cord blood was not the source of EBV infection. The origin of the PTLD was investigated in 5 cases. PTLD was of host origin in 2 patients who failed engraftment and of donor origin in the remaining 3 patients who had complete engraftment. Four of 5 patients with monomorphic PTLD failed to demonstrate significant responses to rituximab and/or reduction of immunosuppression and died within 1 month after diagnosis. The remaining 2 patients with polymorphic PTLD showed complete response to therapy. One patient was alive 35 months after transplant, and the other patient died of infection 6 months after transplant. It is concluded that PTLD arising after UCBT in children occurs early after transplant and represents a serious EBV-related complication. PTLD may be of donor or recipient origin depending on engraftment status. Both monomorphic and polymorphic histology may be seen, and monomorphic histology appears to predict an unfavorable prognosis. |
doi_str_mv | 10.1097/01.pas.0000188030.63706.e7 |
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There were 4 females and 3 males with a median age of 3 years (range, 1-16 years). All 7 patients received UCBT, including 1 patient who received multiple units and 1 transplanted under nonmyeloablative condition. The time interval from UCBT to PTLD averaged 4 months (range, 2 weeks to 9 months). Patients typically presented with high-stage disease with visceral organ involvement. Histology of the PTLDs showed monomorphic morphology in 5 cases and polymorphic morphology in the remaining 2 cases. Bone marrow biopsies were performed in 3 cases and were negative for PTLD. Epstein-Barr virus (EBV) was detected in the PTLD in all 7 patients by in situ hybridization. Evidence of past EBV infection was found in the recipients, but the EBV genome was not detected in the donor cord blood samples, suggesting that donor cord blood was not the source of EBV infection. The origin of the PTLD was investigated in 5 cases. PTLD was of host origin in 2 patients who failed engraftment and of donor origin in the remaining 3 patients who had complete engraftment. Four of 5 patients with monomorphic PTLD failed to demonstrate significant responses to rituximab and/or reduction of immunosuppression and died within 1 month after diagnosis. The remaining 2 patients with polymorphic PTLD showed complete response to therapy. One patient was alive 35 months after transplant, and the other patient died of infection 6 months after transplant. It is concluded that PTLD arising after UCBT in children occurs early after transplant and represents a serious EBV-related complication. PTLD may be of donor or recipient origin depending on engraftment status. Both monomorphic and polymorphic histology may be seen, and monomorphic histology appears to predict an unfavorable prognosis.</description><identifier>ISSN: 0147-5185</identifier><identifier>EISSN: 1532-0979</identifier><identifier>DOI: 10.1097/01.pas.0000188030.63706.e7</identifier><identifier>PMID: 16538052</identifier><identifier>CODEN: AJSPDX</identifier><language>eng</language><publisher>Hagerstown, MD: Lippincott Williams & Wilkins</publisher><subject>Adolescent ; Biological and medical sciences ; Child ; Child, Preschool ; Cord Blood Stem Cell Transplantation - adverse effects ; Epstein-Barr Virus Infections - epidemiology ; Female ; Flow Cytometry ; Hematologic and hematopoietic diseases ; Herpesvirus 4, Human - isolation & purification ; Humans ; Immunohistochemistry ; In Situ Hybridization, Fluorescence ; Infant ; Investigative techniques, diagnostic techniques (general aspects) ; Leukemias. Malignant lymphomas. Malignant reticulosis. Myelofibrosis ; Lymphoproliferative Disorders - etiology ; Lymphoproliferative Disorders - pathology ; Male ; Medical sciences ; Pathology. Cytology. Biochemistry. Spectrometry. Miscellaneous investigative techniques ; Polymerase Chain Reaction ; Ribosomal Proteins - analysis ; RNA-Binding Proteins - analysis</subject><ispartof>The American journal of surgical pathology, 2006-03, Vol.30 (3), p.328-336</ispartof><rights>2006 INIST-CNRS</rights><lds50>peer_reviewed</lds50><woscitedreferencessubscribed>false</woscitedreferencessubscribed><citedby>FETCH-LOGICAL-c413t-bf677b3f9e73c5695a929218f7ecdf1315c33ead7655fd126c19684ce895a3a3</citedby></display><links><openurl>$$Topenurl_article</openurl><openurlfulltext>$$Topenurlfull_article</openurlfulltext><thumbnail>$$Tsyndetics_thumb_exl</thumbnail><link.rule.ids>315,781,785,27929,27930</link.rule.ids><backlink>$$Uhttp://pascal-francis.inist.fr/vibad/index.php?action=getRecordDetail&idt=17614528$$DView record in Pascal Francis$$Hfree_for_read</backlink><backlink>$$Uhttps://www.ncbi.nlm.nih.gov/pubmed/16538052$$D View this record in MEDLINE/PubMed$$Hfree_for_read</backlink></links><search><creatorcontrib>GONG, Jerald Z</creatorcontrib><creatorcontrib>BAYERL, Michael G</creatorcontrib><creatorcontrib>BUCKLEY, Patrick J</creatorcontrib><creatorcontrib>SANDHAUS, Linda M</creatorcontrib><creatorcontrib>SEBASTIAN, Siby</creatorcontrib><creatorcontrib>REHDER, Catherine W</creatorcontrib><creatorcontrib>ROUTBORT, Mark</creatorcontrib><creatorcontrib>LAGOO, Anand S</creatorcontrib><creatorcontrib>SZABOLCS, Paul</creatorcontrib><creatorcontrib>CHIU, Jeanie</creatorcontrib><creatorcontrib>COMITO, Melanie</creatorcontrib><title>Posttransplant lymphoproliferative disorder after umbilical cord blood transplantation in children</title><title>The American journal of surgical pathology</title><addtitle>Am J Surg Pathol</addtitle><description>Reported are 7 cases of posttransplant lymphoproliferative disorder (PTLD) arising in children who received umbilical cord blood transplantation (UCBT). There were 4 females and 3 males with a median age of 3 years (range, 1-16 years). All 7 patients received UCBT, including 1 patient who received multiple units and 1 transplanted under nonmyeloablative condition. The time interval from UCBT to PTLD averaged 4 months (range, 2 weeks to 9 months). Patients typically presented with high-stage disease with visceral organ involvement. Histology of the PTLDs showed monomorphic morphology in 5 cases and polymorphic morphology in the remaining 2 cases. Bone marrow biopsies were performed in 3 cases and were negative for PTLD. Epstein-Barr virus (EBV) was detected in the PTLD in all 7 patients by in situ hybridization. Evidence of past EBV infection was found in the recipients, but the EBV genome was not detected in the donor cord blood samples, suggesting that donor cord blood was not the source of EBV infection. The origin of the PTLD was investigated in 5 cases. PTLD was of host origin in 2 patients who failed engraftment and of donor origin in the remaining 3 patients who had complete engraftment. Four of 5 patients with monomorphic PTLD failed to demonstrate significant responses to rituximab and/or reduction of immunosuppression and died within 1 month after diagnosis. The remaining 2 patients with polymorphic PTLD showed complete response to therapy. One patient was alive 35 months after transplant, and the other patient died of infection 6 months after transplant. It is concluded that PTLD arising after UCBT in children occurs early after transplant and represents a serious EBV-related complication. PTLD may be of donor or recipient origin depending on engraftment status. Both monomorphic and polymorphic histology may be seen, and monomorphic histology appears to predict an unfavorable prognosis.</description><subject>Adolescent</subject><subject>Biological and medical sciences</subject><subject>Child</subject><subject>Child, Preschool</subject><subject>Cord Blood Stem Cell Transplantation - adverse effects</subject><subject>Epstein-Barr Virus Infections - epidemiology</subject><subject>Female</subject><subject>Flow Cytometry</subject><subject>Hematologic and hematopoietic diseases</subject><subject>Herpesvirus 4, Human - isolation & purification</subject><subject>Humans</subject><subject>Immunohistochemistry</subject><subject>In Situ Hybridization, Fluorescence</subject><subject>Infant</subject><subject>Investigative techniques, diagnostic techniques (general aspects)</subject><subject>Leukemias. Malignant lymphomas. Malignant reticulosis. Myelofibrosis</subject><subject>Lymphoproliferative Disorders - etiology</subject><subject>Lymphoproliferative Disorders - pathology</subject><subject>Male</subject><subject>Medical sciences</subject><subject>Pathology. Cytology. Biochemistry. Spectrometry. Miscellaneous investigative techniques</subject><subject>Polymerase Chain Reaction</subject><subject>Ribosomal Proteins - analysis</subject><subject>RNA-Binding Proteins - analysis</subject><issn>0147-5185</issn><issn>1532-0979</issn><fulltext>true</fulltext><rsrctype>article</rsrctype><creationdate>2006</creationdate><recordtype>article</recordtype><sourceid>EIF</sourceid><recordid>eNpFkE1LxDAURYMoOo7-BSmC7lrzkuaj7kT8AkEX7kOaJhhJm5p0BP-9UQcmiwTCefddDkLngBvAnbjC0Mw6N7gckBJT3HAqMG-s2EMrYJTUher20QpDK2oGkh2h45w_Ck4kkEN0BJxRiRlZof415mVJespz0NNShe9xfo9zisE7m_Tiv2w1-BzTYFOl3VLuzdj74I0OlSnfVR9iHKpdRJmJU-Wnyrz7MCQ7naADp0O2p9t3jd7u795uH-vnl4en25vn2rRAl7p3XIieus4KahjvmO5IR0A6Yc3ggAIzlFo9CM6YG4BwAx2XrbGyoFTTNbr8jy3lPzc2L2r02dhQOtm4yaqkt4JQWsDrf9CkmHOyTs3Jjzp9K8DqV7DCoIpgtROs_gSr0myNzrZbNv1oh93o1mgBLraAzsWRK16MzztOcGgZkfQH3NiH1Q</recordid><startdate>20060301</startdate><enddate>20060301</enddate><creator>GONG, Jerald Z</creator><creator>BAYERL, Michael G</creator><creator>BUCKLEY, Patrick J</creator><creator>SANDHAUS, Linda M</creator><creator>SEBASTIAN, Siby</creator><creator>REHDER, Catherine W</creator><creator>ROUTBORT, Mark</creator><creator>LAGOO, Anand S</creator><creator>SZABOLCS, Paul</creator><creator>CHIU, Jeanie</creator><creator>COMITO, Melanie</creator><general>Lippincott Williams & Wilkins</general><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>7X8</scope></search><sort><creationdate>20060301</creationdate><title>Posttransplant lymphoproliferative disorder after umbilical cord blood transplantation in children</title><author>GONG, Jerald Z ; BAYERL, Michael G ; BUCKLEY, Patrick J ; SANDHAUS, Linda M ; SEBASTIAN, Siby ; REHDER, Catherine W ; ROUTBORT, Mark ; LAGOO, Anand S ; SZABOLCS, Paul ; CHIU, Jeanie ; COMITO, Melanie</author></sort><facets><frbrtype>5</frbrtype><frbrgroupid>cdi_FETCH-LOGICAL-c413t-bf677b3f9e73c5695a929218f7ecdf1315c33ead7655fd126c19684ce895a3a3</frbrgroupid><rsrctype>articles</rsrctype><prefilter>articles</prefilter><language>eng</language><creationdate>2006</creationdate><topic>Adolescent</topic><topic>Biological and medical sciences</topic><topic>Child</topic><topic>Child, Preschool</topic><topic>Cord Blood Stem Cell Transplantation - adverse effects</topic><topic>Epstein-Barr Virus Infections - epidemiology</topic><topic>Female</topic><topic>Flow Cytometry</topic><topic>Hematologic and hematopoietic diseases</topic><topic>Herpesvirus 4, Human - isolation & purification</topic><topic>Humans</topic><topic>Immunohistochemistry</topic><topic>In Situ Hybridization, Fluorescence</topic><topic>Infant</topic><topic>Investigative techniques, diagnostic techniques (general aspects)</topic><topic>Leukemias. Malignant lymphomas. Malignant reticulosis. Myelofibrosis</topic><topic>Lymphoproliferative Disorders - etiology</topic><topic>Lymphoproliferative Disorders - pathology</topic><topic>Male</topic><topic>Medical sciences</topic><topic>Pathology. Cytology. Biochemistry. Spectrometry. Miscellaneous investigative techniques</topic><topic>Polymerase Chain Reaction</topic><topic>Ribosomal Proteins - analysis</topic><topic>RNA-Binding Proteins - analysis</topic><toplevel>peer_reviewed</toplevel><toplevel>online_resources</toplevel><creatorcontrib>GONG, Jerald Z</creatorcontrib><creatorcontrib>BAYERL, Michael G</creatorcontrib><creatorcontrib>BUCKLEY, Patrick J</creatorcontrib><creatorcontrib>SANDHAUS, Linda M</creatorcontrib><creatorcontrib>SEBASTIAN, Siby</creatorcontrib><creatorcontrib>REHDER, Catherine W</creatorcontrib><creatorcontrib>ROUTBORT, Mark</creatorcontrib><creatorcontrib>LAGOO, Anand S</creatorcontrib><creatorcontrib>SZABOLCS, Paul</creatorcontrib><creatorcontrib>CHIU, Jeanie</creatorcontrib><creatorcontrib>COMITO, Melanie</creatorcontrib><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>MEDLINE - Academic</collection><jtitle>The American journal of surgical pathology</jtitle></facets><delivery><delcategory>Remote Search Resource</delcategory><fulltext>fulltext</fulltext></delivery><addata><au>GONG, Jerald Z</au><au>BAYERL, Michael G</au><au>BUCKLEY, Patrick J</au><au>SANDHAUS, Linda M</au><au>SEBASTIAN, Siby</au><au>REHDER, Catherine W</au><au>ROUTBORT, Mark</au><au>LAGOO, Anand S</au><au>SZABOLCS, Paul</au><au>CHIU, Jeanie</au><au>COMITO, Melanie</au><format>journal</format><genre>article</genre><ristype>JOUR</ristype><atitle>Posttransplant lymphoproliferative disorder after umbilical cord blood transplantation in children</atitle><jtitle>The American journal of surgical pathology</jtitle><addtitle>Am J Surg Pathol</addtitle><date>2006-03-01</date><risdate>2006</risdate><volume>30</volume><issue>3</issue><spage>328</spage><epage>336</epage><pages>328-336</pages><issn>0147-5185</issn><eissn>1532-0979</eissn><coden>AJSPDX</coden><abstract>Reported are 7 cases of posttransplant lymphoproliferative disorder (PTLD) arising in children who received umbilical cord blood transplantation (UCBT). There were 4 females and 3 males with a median age of 3 years (range, 1-16 years). All 7 patients received UCBT, including 1 patient who received multiple units and 1 transplanted under nonmyeloablative condition. The time interval from UCBT to PTLD averaged 4 months (range, 2 weeks to 9 months). Patients typically presented with high-stage disease with visceral organ involvement. Histology of the PTLDs showed monomorphic morphology in 5 cases and polymorphic morphology in the remaining 2 cases. Bone marrow biopsies were performed in 3 cases and were negative for PTLD. Epstein-Barr virus (EBV) was detected in the PTLD in all 7 patients by in situ hybridization. Evidence of past EBV infection was found in the recipients, but the EBV genome was not detected in the donor cord blood samples, suggesting that donor cord blood was not the source of EBV infection. The origin of the PTLD was investigated in 5 cases. PTLD was of host origin in 2 patients who failed engraftment and of donor origin in the remaining 3 patients who had complete engraftment. Four of 5 patients with monomorphic PTLD failed to demonstrate significant responses to rituximab and/or reduction of immunosuppression and died within 1 month after diagnosis. The remaining 2 patients with polymorphic PTLD showed complete response to therapy. One patient was alive 35 months after transplant, and the other patient died of infection 6 months after transplant. It is concluded that PTLD arising after UCBT in children occurs early after transplant and represents a serious EBV-related complication. PTLD may be of donor or recipient origin depending on engraftment status. Both monomorphic and polymorphic histology may be seen, and monomorphic histology appears to predict an unfavorable prognosis.</abstract><cop>Hagerstown, MD</cop><pub>Lippincott Williams & Wilkins</pub><pmid>16538052</pmid><doi>10.1097/01.pas.0000188030.63706.e7</doi><tpages>9</tpages></addata></record> |
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subjects | Adolescent Biological and medical sciences Child Child, Preschool Cord Blood Stem Cell Transplantation - adverse effects Epstein-Barr Virus Infections - epidemiology Female Flow Cytometry Hematologic and hematopoietic diseases Herpesvirus 4, Human - isolation & purification Humans Immunohistochemistry In Situ Hybridization, Fluorescence Infant Investigative techniques, diagnostic techniques (general aspects) Leukemias. Malignant lymphomas. Malignant reticulosis. Myelofibrosis Lymphoproliferative Disorders - etiology Lymphoproliferative Disorders - pathology Male Medical sciences Pathology. Cytology. Biochemistry. Spectrometry. Miscellaneous investigative techniques Polymerase Chain Reaction Ribosomal Proteins - analysis RNA-Binding Proteins - analysis |
title | Posttransplant lymphoproliferative disorder after umbilical cord blood transplantation in children |
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