Unrelated cord blood transplantation for adult-onset EBV-associated T-cell and NK-cell lymphoproliferative disorders
Adult-onset EBV-associated T-cell and NK-cell lymphoproliferative disorders (EBV-T/NK-LPDs) often progress rapidly, and require allogeneic stem cell transplantation early in the course of treatment. Unrelated cord blood transplantation (UCBT) is a readily available option for patients without HLA-ma...
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Veröffentlicht in: | International journal of hematology 2022-06, Vol.115 (6), p.873-881 |
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creator | Onishi, Yasushi Onodera, Koichi Fukuhara, Noriko Kato, Hiroki Ichikawa, Satoshi Fujiwara, Tohru Yokoyama, Hisayuki Yamada-Fujiwara, Minami Harigae, Hideo |
description | Adult-onset EBV-associated T-cell and NK-cell lymphoproliferative disorders (EBV-T/NK-LPDs) often progress rapidly, and require allogeneic stem cell transplantation early in the course of treatment. Unrelated cord blood transplantation (UCBT) is a readily available option for patients without HLA-matched donors. We retrospectively analyzed the outcomes of 12 UCBT in adult patients with chronic active EBV infection (CAEBV,
n
= 8), EBV-positive hemophagocytic lymphohistiocytosis following primary EBV infection (
n
= 2), hydroa vacciniforme-like lymphoproliferative disorder (
n
= 1), and systemic EBV-positive T-cell lymphoma of childhood (STCLC,
n
= 1). The median age at transplantation was 31.5 years (range 19–58). At the median follow-up time for survivors, which was 6.3 years (range 0.3–11.3), 3-year overall survival (OS) rates in all patients and 8 CAEBV patients were 68.2% (95% CI 28.6–88.9) and 83.3% (95% CI 27.3–97.5), respectively. Graft failure occurred in 4 of 8 CAEBV patients, requiring a second UCBT to achieve neutrophil engraftment. The cumulative incidence of grade II–IV acute GVHD was 33.3% (95% CI 9.1–60.4%). The EBV-DNA load became undetectable or very low after UCBT in all cases. UCBT may be a promising treatment option for adult-onset EBV-T/NK-LPDs. |
doi_str_mv | 10.1007/s12185-022-03313-z |
format | Article |
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n
= 8), EBV-positive hemophagocytic lymphohistiocytosis following primary EBV infection (
n
= 2), hydroa vacciniforme-like lymphoproliferative disorder (
n
= 1), and systemic EBV-positive T-cell lymphoma of childhood (STCLC,
n
= 1). The median age at transplantation was 31.5 years (range 19–58). At the median follow-up time for survivors, which was 6.3 years (range 0.3–11.3), 3-year overall survival (OS) rates in all patients and 8 CAEBV patients were 68.2% (95% CI 28.6–88.9) and 83.3% (95% CI 27.3–97.5), respectively. Graft failure occurred in 4 of 8 CAEBV patients, requiring a second UCBT to achieve neutrophil engraftment. The cumulative incidence of grade II–IV acute GVHD was 33.3% (95% CI 9.1–60.4%). The EBV-DNA load became undetectable or very low after UCBT in all cases. UCBT may be a promising treatment option for adult-onset EBV-T/NK-LPDs.</description><identifier>ISSN: 0925-5710</identifier><identifier>EISSN: 1865-3774</identifier><identifier>DOI: 10.1007/s12185-022-03313-z</identifier><identifier>PMID: 35274195</identifier><language>eng</language><publisher>Singapore: Springer Nature Singapore</publisher><subject>Allografts ; Antigens ; Blood ; Children ; Chronic infection ; Cord blood ; Deoxyribonucleic acid ; Disease ; Disorders ; DNA ; Graft rejection ; Graft-versus-host reaction ; Hematology ; Histiocytosis ; Immunoproliferative diseases ; Infections ; Leukocytes (neutrophilic) ; Lymphatic diseases ; Lymphocytes ; Lymphocytes T ; Lymphocytosis ; Lymphoma ; Medical prognosis ; Medicine ; Medicine & Public Health ; Multiple organ dysfunction syndrome ; Neutrophils ; Oncology ; Original Article ; Patients ; Pediatrics ; Remission (Medicine) ; Stem cell transplantation ; Stem cells ; T-cell lymphoma ; Transplantation</subject><ispartof>International journal of hematology, 2022-06, Vol.115 (6), p.873-881</ispartof><rights>Japanese Society of Hematology 2022</rights><rights>2022. Japanese Society of Hematology.</rights><rights>Japanese Society of Hematology 2022.</rights><lds50>peer_reviewed</lds50><woscitedreferencessubscribed>false</woscitedreferencessubscribed><citedby>FETCH-LOGICAL-c399t-c8bf32ade791d62890eb03c1fba5913da270f4e515a699479489e6de416da6b93</citedby><cites>FETCH-LOGICAL-c399t-c8bf32ade791d62890eb03c1fba5913da270f4e515a699479489e6de416da6b93</cites><orcidid>0000-0003-0032-6994</orcidid></display><links><openurl>$$Topenurl_article</openurl><openurlfulltext>$$Topenurlfull_article</openurlfulltext><thumbnail>$$Tsyndetics_thumb_exl</thumbnail><linktopdf>$$Uhttps://link.springer.com/content/pdf/10.1007/s12185-022-03313-z$$EPDF$$P50$$Gspringer$$H</linktopdf><linktohtml>$$Uhttps://link.springer.com/10.1007/s12185-022-03313-z$$EHTML$$P50$$Gspringer$$H</linktohtml><link.rule.ids>314,780,784,27924,27925,41488,42557,51319</link.rule.ids><backlink>$$Uhttps://www.ncbi.nlm.nih.gov/pubmed/35274195$$D View this record in MEDLINE/PubMed$$Hfree_for_read</backlink></links><search><creatorcontrib>Onishi, Yasushi</creatorcontrib><creatorcontrib>Onodera, Koichi</creatorcontrib><creatorcontrib>Fukuhara, Noriko</creatorcontrib><creatorcontrib>Kato, Hiroki</creatorcontrib><creatorcontrib>Ichikawa, Satoshi</creatorcontrib><creatorcontrib>Fujiwara, Tohru</creatorcontrib><creatorcontrib>Yokoyama, Hisayuki</creatorcontrib><creatorcontrib>Yamada-Fujiwara, Minami</creatorcontrib><creatorcontrib>Harigae, Hideo</creatorcontrib><title>Unrelated cord blood transplantation for adult-onset EBV-associated T-cell and NK-cell lymphoproliferative disorders</title><title>International journal of hematology</title><addtitle>Int J Hematol</addtitle><addtitle>Int J Hematol</addtitle><description>Adult-onset EBV-associated T-cell and NK-cell lymphoproliferative disorders (EBV-T/NK-LPDs) often progress rapidly, and require allogeneic stem cell transplantation early in the course of treatment. Unrelated cord blood transplantation (UCBT) is a readily available option for patients without HLA-matched donors. We retrospectively analyzed the outcomes of 12 UCBT in adult patients with chronic active EBV infection (CAEBV,
n
= 8), EBV-positive hemophagocytic lymphohistiocytosis following primary EBV infection (
n
= 2), hydroa vacciniforme-like lymphoproliferative disorder (
n
= 1), and systemic EBV-positive T-cell lymphoma of childhood (STCLC,
n
= 1). The median age at transplantation was 31.5 years (range 19–58). At the median follow-up time for survivors, which was 6.3 years (range 0.3–11.3), 3-year overall survival (OS) rates in all patients and 8 CAEBV patients were 68.2% (95% CI 28.6–88.9) and 83.3% (95% CI 27.3–97.5), respectively. Graft failure occurred in 4 of 8 CAEBV patients, requiring a second UCBT to achieve neutrophil engraftment. The cumulative incidence of grade II–IV acute GVHD was 33.3% (95% CI 9.1–60.4%). The EBV-DNA load became undetectable or very low after UCBT in all cases. UCBT may be a promising treatment option for adult-onset EBV-T/NK-LPDs.</description><subject>Allografts</subject><subject>Antigens</subject><subject>Blood</subject><subject>Children</subject><subject>Chronic infection</subject><subject>Cord blood</subject><subject>Deoxyribonucleic acid</subject><subject>Disease</subject><subject>Disorders</subject><subject>DNA</subject><subject>Graft rejection</subject><subject>Graft-versus-host reaction</subject><subject>Hematology</subject><subject>Histiocytosis</subject><subject>Immunoproliferative diseases</subject><subject>Infections</subject><subject>Leukocytes (neutrophilic)</subject><subject>Lymphatic diseases</subject><subject>Lymphocytes</subject><subject>Lymphocytes T</subject><subject>Lymphocytosis</subject><subject>Lymphoma</subject><subject>Medical prognosis</subject><subject>Medicine</subject><subject>Medicine & Public Health</subject><subject>Multiple organ dysfunction syndrome</subject><subject>Neutrophils</subject><subject>Oncology</subject><subject>Original Article</subject><subject>Patients</subject><subject>Pediatrics</subject><subject>Remission (Medicine)</subject><subject>Stem cell transplantation</subject><subject>Stem cells</subject><subject>T-cell lymphoma</subject><subject>Transplantation</subject><issn>0925-5710</issn><issn>1865-3774</issn><fulltext>true</fulltext><rsrctype>article</rsrctype><creationdate>2022</creationdate><recordtype>article</recordtype><sourceid>ABUWG</sourceid><sourceid>AFKRA</sourceid><sourceid>BENPR</sourceid><sourceid>CCPQU</sourceid><recordid>eNp9kU1LHTEUhkNpqVfbP9CFBLrpJpqPyWSyVLEqSrvRbkNmcqYdyU1uk4ygv95cx1pw4SoH8n7Bg9AXRg8YpeowM846SSjnhArBBHl4h1asayURSjXv0YpqLolUjO6g3ZxvKWWKNuoj2hGSq4ZpuULlJiTwtoDDQ0wO9z5Gh0uyIW-8DcWWKQY8xoStm30hMWQo-PT4F7E5x2F6cl6TAbzHNjj843K5_f168yduUvTTCKmm3AF2U64VkPIn9GG0PsPn53cP3Xw_vT45J1c_zy5Ojq7IILQuZOj6UXDrQGnmWt5pCj0VAxt7KzUTznJFxwYkk7bVulG66TS0DhrWOtv2Wuyhb0tu3fF3hlzMesrbeTZAnLPhregUk5qLKv36Snob5xTqOrMtpg3nehvIF9WQYs4JRrNJ09qme8Oo2TIxCxNTmZgnJuahmvafo-d-De7F8g9CFYhFkOtX-A3pf_cbsY923JjS</recordid><startdate>20220601</startdate><enddate>20220601</enddate><creator>Onishi, Yasushi</creator><creator>Onodera, Koichi</creator><creator>Fukuhara, Noriko</creator><creator>Kato, Hiroki</creator><creator>Ichikawa, Satoshi</creator><creator>Fujiwara, Tohru</creator><creator>Yokoyama, Hisayuki</creator><creator>Yamada-Fujiwara, Minami</creator><creator>Harigae, Hideo</creator><general>Springer Nature Singapore</general><general>Springer Nature 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prognosis</topic><topic>Medicine</topic><topic>Medicine & Public Health</topic><topic>Multiple organ dysfunction syndrome</topic><topic>Neutrophils</topic><topic>Oncology</topic><topic>Original Article</topic><topic>Patients</topic><topic>Pediatrics</topic><topic>Remission (Medicine)</topic><topic>Stem cell transplantation</topic><topic>Stem cells</topic><topic>T-cell lymphoma</topic><topic>Transplantation</topic><toplevel>peer_reviewed</toplevel><toplevel>online_resources</toplevel><creatorcontrib>Onishi, Yasushi</creatorcontrib><creatorcontrib>Onodera, Koichi</creatorcontrib><creatorcontrib>Fukuhara, Noriko</creatorcontrib><creatorcontrib>Kato, Hiroki</creatorcontrib><creatorcontrib>Ichikawa, Satoshi</creatorcontrib><creatorcontrib>Fujiwara, Tohru</creatorcontrib><creatorcontrib>Yokoyama, Hisayuki</creatorcontrib><creatorcontrib>Yamada-Fujiwara, Minami</creatorcontrib><creatorcontrib>Harigae, 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Academic</collection><jtitle>International journal of hematology</jtitle></facets><delivery><delcategory>Remote Search Resource</delcategory><fulltext>fulltext</fulltext></delivery><addata><au>Onishi, Yasushi</au><au>Onodera, Koichi</au><au>Fukuhara, Noriko</au><au>Kato, Hiroki</au><au>Ichikawa, Satoshi</au><au>Fujiwara, Tohru</au><au>Yokoyama, Hisayuki</au><au>Yamada-Fujiwara, Minami</au><au>Harigae, Hideo</au><format>journal</format><genre>article</genre><ristype>JOUR</ristype><atitle>Unrelated cord blood transplantation for adult-onset EBV-associated T-cell and NK-cell lymphoproliferative disorders</atitle><jtitle>International journal of hematology</jtitle><stitle>Int J Hematol</stitle><addtitle>Int J Hematol</addtitle><date>2022-06-01</date><risdate>2022</risdate><volume>115</volume><issue>6</issue><spage>873</spage><epage>881</epage><pages>873-881</pages><issn>0925-5710</issn><eissn>1865-3774</eissn><abstract>Adult-onset EBV-associated T-cell and NK-cell lymphoproliferative disorders (EBV-T/NK-LPDs) often progress rapidly, and require allogeneic stem cell transplantation early in the course of treatment. Unrelated cord blood transplantation (UCBT) is a readily available option for patients without HLA-matched donors. We retrospectively analyzed the outcomes of 12 UCBT in adult patients with chronic active EBV infection (CAEBV,
n
= 8), EBV-positive hemophagocytic lymphohistiocytosis following primary EBV infection (
n
= 2), hydroa vacciniforme-like lymphoproliferative disorder (
n
= 1), and systemic EBV-positive T-cell lymphoma of childhood (STCLC,
n
= 1). The median age at transplantation was 31.5 years (range 19–58). At the median follow-up time for survivors, which was 6.3 years (range 0.3–11.3), 3-year overall survival (OS) rates in all patients and 8 CAEBV patients were 68.2% (95% CI 28.6–88.9) and 83.3% (95% CI 27.3–97.5), respectively. Graft failure occurred in 4 of 8 CAEBV patients, requiring a second UCBT to achieve neutrophil engraftment. The cumulative incidence of grade II–IV acute GVHD was 33.3% (95% CI 9.1–60.4%). The EBV-DNA load became undetectable or very low after UCBT in all cases. UCBT may be a promising treatment option for adult-onset EBV-T/NK-LPDs.</abstract><cop>Singapore</cop><pub>Springer Nature Singapore</pub><pmid>35274195</pmid><doi>10.1007/s12185-022-03313-z</doi><tpages>9</tpages><orcidid>https://orcid.org/0000-0003-0032-6994</orcidid></addata></record> |
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subjects | Allografts Antigens Blood Children Chronic infection Cord blood Deoxyribonucleic acid Disease Disorders DNA Graft rejection Graft-versus-host reaction Hematology Histiocytosis Immunoproliferative diseases Infections Leukocytes (neutrophilic) Lymphatic diseases Lymphocytes Lymphocytes T Lymphocytosis Lymphoma Medical prognosis Medicine Medicine & Public Health Multiple organ dysfunction syndrome Neutrophils Oncology Original Article Patients Pediatrics Remission (Medicine) Stem cell transplantation Stem cells T-cell lymphoma Transplantation |
title | Unrelated cord blood transplantation for adult-onset EBV-associated T-cell and NK-cell lymphoproliferative disorders |
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