Malignancy‐associated haemophagocytic lymphohistiocytosis in children and adolescents
Summary Haemophagocytic lymphohistiocytosis (HLH) in the context of malignancy is mainly considered a challenge of adult haematology. While this association is also observed in children, little is known regarding inciting factors, appropriate treatment and prognosis. We retrospectively analysed 29 p...
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Veröffentlicht in: | British journal of haematology 2015-08, Vol.170 (4), p.539-549 |
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creator | Lehmberg, Kai Sprekels, Björn Nichols, Kim E. Woessmann, Wilhelm Müller, Ingo Suttorp, Meinolf Bernig, Toralf Beutel, Karin Bode, Sebastian F. N. Kentouche, Karim Kolb, Reinhard Längler, Alfred Minkov, Milen Schilling, Freimut H. Schmid, Irene Vieth, Simon Ehl, Stephan Zur Stadt, Udo Janka, Gritta E. |
description | Summary
Haemophagocytic lymphohistiocytosis (HLH) in the context of malignancy is mainly considered a challenge of adult haematology. While this association is also observed in children, little is known regarding inciting factors, appropriate treatment and prognosis. We retrospectively analysed 29 paediatric and adolescent patients for presenting features, type of neoplasm or preceding chemotherapy, treatment and outcome. Haemophagocytic lymphohistiocytosis was considered triggered by the malignancy (M‐HLH) in 21 patients, most of whom had T‐ (n = 12) or B‐cell neoplasms (n = 7), with Epstein–Barr virus as a co‐trigger in five patients. In eight patients, HLH occurred during chemotherapy (Ch‐HLH) for malignancy, mainly acute leukaemias (n = 7); an infectious trigger was found in seven. In M‐ and Ch‐HLH, median overall survival was 1·2 and 0·9 years, and the 6 month survival rates were 67% and 63%, respectively. Seven of 11 deceased M‐HLH patients exhibited active malignancy and HLH at the time of death, while only two out of five deceased Ch‐HLH patients had evidence of active HLH. To overcome HLH, malignancy‐ and HLH‐directed treatments were administered in the M‐HLH cohort; however, it was not possible to determine superiority of one approach over the other. For Ch‐HLH, treatment ranged from postponement of chemotherapy to the use of etoposide‐containing regimens. |
doi_str_mv | 10.1111/bjh.13462 |
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Haemophagocytic lymphohistiocytosis (HLH) in the context of malignancy is mainly considered a challenge of adult haematology. While this association is also observed in children, little is known regarding inciting factors, appropriate treatment and prognosis. We retrospectively analysed 29 paediatric and adolescent patients for presenting features, type of neoplasm or preceding chemotherapy, treatment and outcome. Haemophagocytic lymphohistiocytosis was considered triggered by the malignancy (M‐HLH) in 21 patients, most of whom had T‐ (n = 12) or B‐cell neoplasms (n = 7), with Epstein–Barr virus as a co‐trigger in five patients. In eight patients, HLH occurred during chemotherapy (Ch‐HLH) for malignancy, mainly acute leukaemias (n = 7); an infectious trigger was found in seven. In M‐ and Ch‐HLH, median overall survival was 1·2 and 0·9 years, and the 6 month survival rates were 67% and 63%, respectively. Seven of 11 deceased M‐HLH patients exhibited active malignancy and HLH at the time of death, while only two out of five deceased Ch‐HLH patients had evidence of active HLH. To overcome HLH, malignancy‐ and HLH‐directed treatments were administered in the M‐HLH cohort; however, it was not possible to determine superiority of one approach over the other. For Ch‐HLH, treatment ranged from postponement of chemotherapy to the use of etoposide‐containing regimens.</description><identifier>ISSN: 0007-1048</identifier><identifier>EISSN: 1365-2141</identifier><identifier>DOI: 10.1111/bjh.13462</identifier><identifier>PMID: 25940575</identifier><language>eng</language><publisher>England</publisher><subject>Acute Disease ; Adolescent ; Adult ; anaplastic large cell lymphoma ; Child ; Child, Preschool ; Disease-Free Survival ; Epstein-Barr Virus Infections - complications ; Epstein-Barr Virus Infections - drug therapy ; Epstein-Barr Virus Infections - mortality ; Female ; hemophagocytic syndrome ; Hodgkin lymphoma ; Humans ; Infant ; Infant, Newborn ; Leukemia - drug therapy ; Leukemia - mortality ; Lymphohistiocytosis, Hemophagocytic - etiology ; Lymphohistiocytosis, Hemophagocytic - mortality ; Lymphohistiocytosis, Hemophagocytic - therapy ; macrophage activation syndrome ; Male ; Retrospective Studies ; Survival Rate ; T‐cell lymphoma</subject><ispartof>British journal of haematology, 2015-08, Vol.170 (4), p.539-549</ispartof><rights>2015 John Wiley & Sons Ltd</rights><rights>2015 John Wiley & Sons Ltd.</rights><lds50>peer_reviewed</lds50><oa>free_for_read</oa><woscitedreferencessubscribed>false</woscitedreferencessubscribed><citedby>FETCH-LOGICAL-c3602-6a991c6ec44091583374b86a064afd776489779583e21540890ebe3514babe2b3</citedby><cites>FETCH-LOGICAL-c3602-6a991c6ec44091583374b86a064afd776489779583e21540890ebe3514babe2b3</cites></display><links><openurl>$$Topenurl_article</openurl><openurlfulltext>$$Topenurlfull_article</openurlfulltext><thumbnail>$$Tsyndetics_thumb_exl</thumbnail><linktopdf>$$Uhttps://onlinelibrary.wiley.com/doi/pdf/10.1111%2Fbjh.13462$$EPDF$$P50$$Gwiley$$H</linktopdf><linktohtml>$$Uhttps://onlinelibrary.wiley.com/doi/full/10.1111%2Fbjh.13462$$EHTML$$P50$$Gwiley$$H</linktohtml><link.rule.ids>314,780,784,1417,1433,27924,27925,45574,45575,46409,46833</link.rule.ids><backlink>$$Uhttps://www.ncbi.nlm.nih.gov/pubmed/25940575$$D View this record in MEDLINE/PubMed$$Hfree_for_read</backlink></links><search><creatorcontrib>Lehmberg, Kai</creatorcontrib><creatorcontrib>Sprekels, Björn</creatorcontrib><creatorcontrib>Nichols, Kim E.</creatorcontrib><creatorcontrib>Woessmann, Wilhelm</creatorcontrib><creatorcontrib>Müller, Ingo</creatorcontrib><creatorcontrib>Suttorp, Meinolf</creatorcontrib><creatorcontrib>Bernig, Toralf</creatorcontrib><creatorcontrib>Beutel, Karin</creatorcontrib><creatorcontrib>Bode, Sebastian F. N.</creatorcontrib><creatorcontrib>Kentouche, Karim</creatorcontrib><creatorcontrib>Kolb, Reinhard</creatorcontrib><creatorcontrib>Längler, Alfred</creatorcontrib><creatorcontrib>Minkov, Milen</creatorcontrib><creatorcontrib>Schilling, Freimut H.</creatorcontrib><creatorcontrib>Schmid, Irene</creatorcontrib><creatorcontrib>Vieth, Simon</creatorcontrib><creatorcontrib>Ehl, Stephan</creatorcontrib><creatorcontrib>Zur Stadt, Udo</creatorcontrib><creatorcontrib>Janka, Gritta E.</creatorcontrib><title>Malignancy‐associated haemophagocytic lymphohistiocytosis in children and adolescents</title><title>British journal of haematology</title><addtitle>Br J Haematol</addtitle><description>Summary
Haemophagocytic lymphohistiocytosis (HLH) in the context of malignancy is mainly considered a challenge of adult haematology. While this association is also observed in children, little is known regarding inciting factors, appropriate treatment and prognosis. We retrospectively analysed 29 paediatric and adolescent patients for presenting features, type of neoplasm or preceding chemotherapy, treatment and outcome. Haemophagocytic lymphohistiocytosis was considered triggered by the malignancy (M‐HLH) in 21 patients, most of whom had T‐ (n = 12) or B‐cell neoplasms (n = 7), with Epstein–Barr virus as a co‐trigger in five patients. In eight patients, HLH occurred during chemotherapy (Ch‐HLH) for malignancy, mainly acute leukaemias (n = 7); an infectious trigger was found in seven. In M‐ and Ch‐HLH, median overall survival was 1·2 and 0·9 years, and the 6 month survival rates were 67% and 63%, respectively. Seven of 11 deceased M‐HLH patients exhibited active malignancy and HLH at the time of death, while only two out of five deceased Ch‐HLH patients had evidence of active HLH. To overcome HLH, malignancy‐ and HLH‐directed treatments were administered in the M‐HLH cohort; however, it was not possible to determine superiority of one approach over the other. For Ch‐HLH, treatment ranged from postponement of chemotherapy to the use of etoposide‐containing regimens.</description><subject>Acute Disease</subject><subject>Adolescent</subject><subject>Adult</subject><subject>anaplastic large cell lymphoma</subject><subject>Child</subject><subject>Child, Preschool</subject><subject>Disease-Free Survival</subject><subject>Epstein-Barr Virus Infections - complications</subject><subject>Epstein-Barr Virus Infections - drug therapy</subject><subject>Epstein-Barr Virus Infections - mortality</subject><subject>Female</subject><subject>hemophagocytic syndrome</subject><subject>Hodgkin lymphoma</subject><subject>Humans</subject><subject>Infant</subject><subject>Infant, Newborn</subject><subject>Leukemia - drug therapy</subject><subject>Leukemia - mortality</subject><subject>Lymphohistiocytosis, Hemophagocytic - etiology</subject><subject>Lymphohistiocytosis, Hemophagocytic - mortality</subject><subject>Lymphohistiocytosis, Hemophagocytic - therapy</subject><subject>macrophage activation syndrome</subject><subject>Male</subject><subject>Retrospective Studies</subject><subject>Survival Rate</subject><subject>T‐cell lymphoma</subject><issn>0007-1048</issn><issn>1365-2141</issn><fulltext>true</fulltext><rsrctype>article</rsrctype><creationdate>2015</creationdate><recordtype>article</recordtype><sourceid>EIF</sourceid><recordid>eNp1kLtOwzAUQC0EoqUw8AMoIwxpr-NXMkIFFFTEAmK0HMdtXCVxiFOhbHwC38iXkBJgw8uVfI-Org5CpximuH-zdJNPMaE82kNjTDgLI0zxPhoDgAgx0HiEjrzfAGACDB-iUcQSCkywMXp5UIVdV6rS3ef7h_LeaatakwW5MqWrc7V2umutDoqurHOXW9_a3Y_z1ge2CnRui6wxVaCqLFCZK4zXpmr9MTpYqcKbk585Qc8310_zRbh8vL2bXy5DTThEIVdJgjU3mlJIMIsJETSNuQJO1SoTgtM4ESLpFybCjEKcgEkNYZimKjVRSibofPDWjXvdGt_K0vYXFIWqjNt6iQVEEHNBaI9eDKhunPeNWcm6saVqOolB7jrKvqP87tizZz_abVqa7I_8DdcDswF4s4Xp_jfJq_vFoPwCj1B9sQ</recordid><startdate>201508</startdate><enddate>201508</enddate><creator>Lehmberg, Kai</creator><creator>Sprekels, Björn</creator><creator>Nichols, Kim E.</creator><creator>Woessmann, Wilhelm</creator><creator>Müller, Ingo</creator><creator>Suttorp, Meinolf</creator><creator>Bernig, Toralf</creator><creator>Beutel, Karin</creator><creator>Bode, Sebastian F. N.</creator><creator>Kentouche, Karim</creator><creator>Kolb, Reinhard</creator><creator>Längler, Alfred</creator><creator>Minkov, Milen</creator><creator>Schilling, Freimut H.</creator><creator>Schmid, Irene</creator><creator>Vieth, Simon</creator><creator>Ehl, Stephan</creator><creator>Zur Stadt, Udo</creator><creator>Janka, Gritta E.</creator><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>201508</creationdate><title>Malignancy‐associated haemophagocytic lymphohistiocytosis in children and adolescents</title><author>Lehmberg, Kai ; Sprekels, Björn ; Nichols, Kim E. ; Woessmann, Wilhelm ; Müller, Ingo ; Suttorp, Meinolf ; Bernig, Toralf ; Beutel, Karin ; Bode, Sebastian F. N. ; Kentouche, Karim ; Kolb, Reinhard ; Längler, Alfred ; Minkov, Milen ; Schilling, Freimut H. ; Schmid, Irene ; Vieth, Simon ; Ehl, Stephan ; Zur Stadt, Udo ; Janka, Gritta E.</author></sort><facets><frbrtype>5</frbrtype><frbrgroupid>cdi_FETCH-LOGICAL-c3602-6a991c6ec44091583374b86a064afd776489779583e21540890ebe3514babe2b3</frbrgroupid><rsrctype>articles</rsrctype><prefilter>articles</prefilter><language>eng</language><creationdate>2015</creationdate><topic>Acute Disease</topic><topic>Adolescent</topic><topic>Adult</topic><topic>anaplastic large cell lymphoma</topic><topic>Child</topic><topic>Child, Preschool</topic><topic>Disease-Free Survival</topic><topic>Epstein-Barr Virus Infections - complications</topic><topic>Epstein-Barr Virus Infections - drug therapy</topic><topic>Epstein-Barr Virus Infections - mortality</topic><topic>Female</topic><topic>hemophagocytic syndrome</topic><topic>Hodgkin lymphoma</topic><topic>Humans</topic><topic>Infant</topic><topic>Infant, Newborn</topic><topic>Leukemia - drug therapy</topic><topic>Leukemia - mortality</topic><topic>Lymphohistiocytosis, Hemophagocytic - etiology</topic><topic>Lymphohistiocytosis, Hemophagocytic - mortality</topic><topic>Lymphohistiocytosis, Hemophagocytic - therapy</topic><topic>macrophage activation syndrome</topic><topic>Male</topic><topic>Retrospective Studies</topic><topic>Survival Rate</topic><topic>T‐cell lymphoma</topic><toplevel>peer_reviewed</toplevel><toplevel>online_resources</toplevel><creatorcontrib>Lehmberg, Kai</creatorcontrib><creatorcontrib>Sprekels, Björn</creatorcontrib><creatorcontrib>Nichols, Kim E.</creatorcontrib><creatorcontrib>Woessmann, Wilhelm</creatorcontrib><creatorcontrib>Müller, Ingo</creatorcontrib><creatorcontrib>Suttorp, Meinolf</creatorcontrib><creatorcontrib>Bernig, Toralf</creatorcontrib><creatorcontrib>Beutel, Karin</creatorcontrib><creatorcontrib>Bode, Sebastian F. N.</creatorcontrib><creatorcontrib>Kentouche, Karim</creatorcontrib><creatorcontrib>Kolb, Reinhard</creatorcontrib><creatorcontrib>Längler, Alfred</creatorcontrib><creatorcontrib>Minkov, Milen</creatorcontrib><creatorcontrib>Schilling, Freimut H.</creatorcontrib><creatorcontrib>Schmid, Irene</creatorcontrib><creatorcontrib>Vieth, Simon</creatorcontrib><creatorcontrib>Ehl, Stephan</creatorcontrib><creatorcontrib>Zur Stadt, Udo</creatorcontrib><creatorcontrib>Janka, Gritta E.</creatorcontrib><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>British journal of haematology</jtitle></facets><delivery><delcategory>Remote Search Resource</delcategory><fulltext>fulltext</fulltext></delivery><addata><au>Lehmberg, Kai</au><au>Sprekels, Björn</au><au>Nichols, Kim E.</au><au>Woessmann, Wilhelm</au><au>Müller, Ingo</au><au>Suttorp, Meinolf</au><au>Bernig, Toralf</au><au>Beutel, Karin</au><au>Bode, Sebastian F. N.</au><au>Kentouche, Karim</au><au>Kolb, Reinhard</au><au>Längler, Alfred</au><au>Minkov, Milen</au><au>Schilling, Freimut H.</au><au>Schmid, Irene</au><au>Vieth, Simon</au><au>Ehl, Stephan</au><au>Zur Stadt, Udo</au><au>Janka, Gritta E.</au><format>journal</format><genre>article</genre><ristype>JOUR</ristype><atitle>Malignancy‐associated haemophagocytic lymphohistiocytosis in children and adolescents</atitle><jtitle>British journal of haematology</jtitle><addtitle>Br J Haematol</addtitle><date>2015-08</date><risdate>2015</risdate><volume>170</volume><issue>4</issue><spage>539</spage><epage>549</epage><pages>539-549</pages><issn>0007-1048</issn><eissn>1365-2141</eissn><abstract>Summary
Haemophagocytic lymphohistiocytosis (HLH) in the context of malignancy is mainly considered a challenge of adult haematology. While this association is also observed in children, little is known regarding inciting factors, appropriate treatment and prognosis. We retrospectively analysed 29 paediatric and adolescent patients for presenting features, type of neoplasm or preceding chemotherapy, treatment and outcome. Haemophagocytic lymphohistiocytosis was considered triggered by the malignancy (M‐HLH) in 21 patients, most of whom had T‐ (n = 12) or B‐cell neoplasms (n = 7), with Epstein–Barr virus as a co‐trigger in five patients. In eight patients, HLH occurred during chemotherapy (Ch‐HLH) for malignancy, mainly acute leukaemias (n = 7); an infectious trigger was found in seven. In M‐ and Ch‐HLH, median overall survival was 1·2 and 0·9 years, and the 6 month survival rates were 67% and 63%, respectively. Seven of 11 deceased M‐HLH patients exhibited active malignancy and HLH at the time of death, while only two out of five deceased Ch‐HLH patients had evidence of active HLH. To overcome HLH, malignancy‐ and HLH‐directed treatments were administered in the M‐HLH cohort; however, it was not possible to determine superiority of one approach over the other. For Ch‐HLH, treatment ranged from postponement of chemotherapy to the use of etoposide‐containing regimens.</abstract><cop>England</cop><pmid>25940575</pmid><doi>10.1111/bjh.13462</doi><tpages>11</tpages><oa>free_for_read</oa></addata></record> |
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subjects | Acute Disease Adolescent Adult anaplastic large cell lymphoma Child Child, Preschool Disease-Free Survival Epstein-Barr Virus Infections - complications Epstein-Barr Virus Infections - drug therapy Epstein-Barr Virus Infections - mortality Female hemophagocytic syndrome Hodgkin lymphoma Humans Infant Infant, Newborn Leukemia - drug therapy Leukemia - mortality Lymphohistiocytosis, Hemophagocytic - etiology Lymphohistiocytosis, Hemophagocytic - mortality Lymphohistiocytosis, Hemophagocytic - therapy macrophage activation syndrome Male Retrospective Studies Survival Rate T‐cell lymphoma |
title | Malignancy‐associated haemophagocytic lymphohistiocytosis in children and adolescents |
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