EBV-driven lymphoid neoplasms associated with pediatric ALL
maintenance therapy

J 2023

EBV-driven lymphoid neoplasms associated with pediatric ALL maintenance therapy

ELITZUR, Sarah, Ajay VORA, Birgit BURKHARDT, Hiroto INABA, Andishe ATTARBASCHI et. al.

Basic information

Original name

EBV-driven lymphoid neoplasms associated with pediatric ALL maintenance therapy

Authors

ELITZUR, Sarah (guarantor), Ajay VORA, Birgit BURKHARDT, Hiroto INABA, Andishe ATTARBASCHI, Andre BARUCHEL, Gabriele ESCHERICH, Brenda GIBSON, Hsi-Che LIU, Mignon LOH, V Moorman ANTHONY, Anja MOERICKE, Rob PIETERS, Anne UYTTEBROECK, Susan BAIRD, Jack BARTRAM, Shlomit BARZILAI-BIRENBOIM, Sandeep BATRA, Miriam BEN-HAROSH, Yves BERTRAND, Trudy BUITENKAMP, Kenneth CALDWELL, Ricardo DRUT, Ashley V GEERLINKS, Gil GILAD, John GRAINGER, Stephanie HAOUY, Nicholas HEANEY, Mary HUANG, Danielle INGHAM, Zdenka KŘENOVÁ (203 Czech Republic, belonging to the institution), Michaela KUHLEN, Thomas LEHRNBECHER, Atsushi MANABE, Felix NIGGLI, Claudia PARIS, Shoshana REVEL-VILK, Pierre ROHRLICH, Mohamad G SINNO, Tomasz SZCZEPANSKI, Melanie TAMESBERGER, Rajasekharan WARRIER, Matthias WOLFL, Ronit NIREL, Shai IZRAELI, Arndt BORKHARDT and Kjeld SCHMIEGELOW

Edition

Blood, Washington DC, USA, American Society of Hematology, 2023, 0006-4971

Other information

Language

English

Type of outcome

Článek v odborném periodiku

Field of Study

30204 Oncology

Country of publisher

United States of America

Confidentiality degree

není předmětem státního či obchodního tajemství

References:

URL

Impact factor

Impact factor: 20.300 in 2022

RIV identification code

RIV/00216224:14110/23:00131032

Organization unit

Faculty of Medicine

DOI

http://dx.doi.org/10.1182/blood.2022016975

UT WoS

000968991200001

Keywords in English

EBV-driven lymphoid neoplasms; pediatric ALL; maintenance therapy

Abstract

V originále

The development of a second malignancy after the diagnosis of childhood acute lymphoblastic leukemia (ALL) is a rare event. Certain second malignancies have been linked with specific elements of leukemia therapy, yet the etiology of most second neoplasms remains obscure and their optimal management strategies are unclear. This is a first comprehensive report of non-Hodgkin lymphomas (NHLs) following pediatric ALL therapy, excluding stem-cell transplantation. We analyzed data of patients who developed NHL following ALL diagnosis and were enrolled in 12 collaborative pediatric ALL trials between 1980-2018. Eighty-five patients developed NHL, with mature B-cell lymphoproliferations as the dominant subtype (56 of 85 cases). Forty-six of these 56 cases (82%) occurred during or within 6 months of maintenance therapy. The majority exhibited histopathological characteristics associated with immunodeficiency (65%), predominantly evidence of Epstein-Barr virus-driven lymphoproliferation. We investigated 66 cases of post-ALL immunodeficiency-associated lymphoid neoplasms, 52 from our study and 14 additional cases from a literature search. With a median follow-up of 4.9 years, the 5-year overall survival for the 66 patients with immunodeficiency-associated lymphoid neoplasms was 67.4% (95% confidence interval [CI], 56-81). Five-year cumulative risks of lymphoid neoplasm- and leukemia-related mortality were 20% (95% CI, 10.2-30) and 12.4% (95% CI, 2.7-22), respectively. Concurrent hemophagocytic lymphohistiocytosis was associated with increased mortality (hazard ratio, 7.32; 95% CI, 1.62-32.98; P =.01). A large proportion of post-ALL lymphoid neoplasms are associated with an immunodeficient state, likely precipitated by ALL maintenance therapy. Awareness of this underrecognized entity and pertinent diagnostic tests are crucial for early diagnosis and optimal therapy.

Links

MUNI/A/1395/2022, interní kód MU

Name: Personalizovaná léčba v dětské onkologii: multimodální theranostický přístup a „N-of-1 clinical trials“

Investor: Masaryk University

Citovat

ELITZUR, Sarah, Ajay VORA, Birgit BURKHARDT, Hiroto INABA, Andishe ATTARBASCHI, Andre BARUCHEL, Gabriele ESCHERICH, Brenda GIBSON, Hsi-Che LIU, Mignon LOH, V Moorman ANTHONY, Anja MOERICKE, Rob PIETERS, Anne UYTTEBROECK, Susan BAIRD, Jack BARTRAM, Shlomit BARZILAI-BIRENBOIM, Sandeep BATRA, Miriam BEN-HAROSH, Yves BERTRAND, Trudy BUITENKAMP, Kenneth CALDWELL, Ricardo DRUT, Ashley V GEERLINKS, Gil GILAD, John GRAINGER, Stephanie HAOUY, Nicholas HEANEY, Mary HUANG, Danielle INGHAM, Zdenka KŘENOVÁ, Michaela KUHLEN, Thomas LEHRNBECHER, Atsushi MANABE, Felix NIGGLI, Claudia PARIS, Shoshana REVEL-VILK, Pierre ROHRLICH, Mohamad G SINNO, Tomasz SZCZEPANSKI, Melanie TAMESBERGER, Rajasekharan WARRIER, Matthias WOLFL, Ronit NIREL, Shai IZRAELI, Arndt BORKHARDT and Kjeld SCHMIEGELOW. EBV-driven lymphoid neoplasms associated with pediatric ALL maintenance therapy. Blood. Washington DC, USA: American Society of Hematology, 2023, vol. 141, No 7, p. 743-755. ISSN 0006-4971. Available from: https://dx.doi.org/10.1182/blood.2022016975.

@article{2293058,
   author = {Elitzur, Sarah and Vora, Ajay and Burkhardt, Birgit and Inaba, Hiroto and Attarbaschi, Andishe and Baruchel, Andre and Escherich, Gabriele and Gibson, Brenda and Liu, HsiandChe and Loh, Mignon and Anthony, V Moorman and Moericke, Anja and Pieters, Rob and Uyttebroeck, Anne and Baird, Susan and Bartram, Jack and BarzilaiandBirenboim, Shlomit and Batra, Sandeep and BenandHarosh, Miriam and Bertrand, Yves and Buitenkamp, Trudy and Caldwell, Kenneth and Drut, Ricardo and Geerlinks, Ashley V and Gilad, Gil and Grainger, John and Haouy, Stephanie and Heaney, Nicholas and Huang, Mary and Ingham, Danielle and Křenová, Zdenka and Kuhlen, Michaela and Lehrnbecher, Thomas and Manabe, Atsushi and Niggli, Felix and Paris, Claudia and RevelandVilk, Shoshana and Rohrlich, Pierre and Sinno, Mohamad G and Szczepanski, Tomasz and Tamesberger, Melanie and Warrier, Rajasekharan and Wolfl, Matthias and Nirel, Ronit and Izraeli, Shai and Borkhardt, Arndt and Schmiegelow, Kjeld},
   article_location = {Washington DC, USA},
   article_number = {7},
   doi = {http://dx.doi.org/10.1182/blood.2022016975},
   keywords = {EBV-driven lymphoid neoplasms; pediatric ALL; maintenance therapy},
   language = {eng},
   issn = {0006-4971},
   journal = {Blood},
   title = {EBV-driven lymphoid neoplasms associated with pediatric ALL maintenance therapy},
   url = {https://ashpublications.org/blood/article-abstract/141/7/743/487024/EBV-driven-lymphoid-neoplasms-associated-with?redirectedFrom=fulltext},
   volume = {141},
   year = {2023}
}

TY  - JOUR
ID  - 2293058
AU  - Elitzur, Sarah - Vora, Ajay - Burkhardt, Birgit - Inaba, Hiroto - Attarbaschi, Andishe - Baruchel, Andre - Escherich, Gabriele - Gibson, Brenda - Liu, Hsi-Che - Loh, Mignon - Anthony, V Moorman - Moericke, Anja - Pieters, Rob - Uyttebroeck, Anne - Baird, Susan - Bartram, Jack - Barzilai-Birenboim, Shlomit - Batra, Sandeep - Ben-Harosh, Miriam - Bertrand, Yves - Buitenkamp, Trudy - Caldwell, Kenneth - Drut, Ricardo - Geerlinks, Ashley V - Gilad, Gil - Grainger, John - Haouy, Stephanie - Heaney, Nicholas - Huang, Mary - Ingham, Danielle - Křenová, Zdenka - Kuhlen, Michaela - Lehrnbecher, Thomas - Manabe, Atsushi - Niggli, Felix - Paris, Claudia - Revel-Vilk, Shoshana - Rohrlich, Pierre - Sinno, Mohamad G - Szczepanski, Tomasz - Tamesberger, Melanie - Warrier, Rajasekharan - Wolfl, Matthias - Nirel, Ronit - Izraeli, Shai - Borkhardt, Arndt - Schmiegelow, Kjeld
PY  - 2023
TI  - EBV-driven lymphoid neoplasms associated with pediatric ALL maintenance therapy
JF  - Blood
VL  - 141
IS  - 7
SP  - 743-755
EP  - 743-755
PB  - American Society of Hematology
SN  - 00064971
KW  - EBV-driven lymphoid neoplasms
KW  - pediatric ALL
KW  - maintenance therapy
UR  - https://ashpublications.org/blood/article-abstract/141/7/743/487024/EBV-driven-lymphoid-neoplasms-associated-with?redirectedFrom=fulltext
N2  - The development of a second malignancy after the diagnosis of childhood acute lymphoblastic leukemia (ALL) is a rare event. Certain second malignancies have been linked with specific elements of leukemia therapy, yet the etiology of most second neoplasms remains obscure and their optimal management strategies are unclear. This is a first comprehensive report of non-Hodgkin lymphomas (NHLs) following pediatric ALL therapy, excluding stem-cell transplantation. We analyzed data of patients who developed NHL following ALL diagnosis and were enrolled in 12 collaborative pediatric ALL trials between 1980-2018. Eighty-five patients developed NHL, with mature B-cell lymphoproliferations as the dominant subtype (56 of 85 cases). Forty-six of these 56 cases (82%) occurred during or within 6 months of maintenance therapy. The majority exhibited histopathological characteristics associated with immunodeficiency (65%), predominantly evidence of Epstein-Barr virus-driven lymphoproliferation. We investigated 66 cases of post-ALL immunodeficiency-associated lymphoid neoplasms, 52 from our study and 14 additional cases from a literature search. With a median follow-up of 4.9 years, the 5-year overall survival for the 66 patients with immunodeficiency-associated lymphoid neoplasms was 67.4% (95% confidence interval [CI], 56-81). Five-year cumulative risks of lymphoid neoplasm- and leukemia-related mortality were 20% (95% CI, 10.2-30) and 12.4% (95% CI, 2.7-22), respectively. Concurrent hemophagocytic lymphohistiocytosis was associated with increased mortality (hazard ratio, 7.32; 95% CI, 1.62-32.98; P =.01). A large proportion of post-ALL lymphoid neoplasms are associated with an immunodeficient state, likely precipitated by ALL maintenance therapy. Awareness of this underrecognized entity and pertinent diagnostic tests are crucial for early diagnosis and optimal therapy.
ER  -

ELITZUR, Sarah, Ajay VORA, Birgit BURKHARDT, Hiroto INABA, Andishe ATTARBASCHI, Andre BARUCHEL, Gabriele ESCHERICH, Brenda GIBSON, Hsi-Che LIU, Mignon LOH, V Moorman ANTHONY, Anja MOERICKE, Rob PIETERS, Anne UYTTEBROECK, Susan BAIRD, Jack BARTRAM, Shlomit BARZILAI-BIRENBOIM, Sandeep BATRA, Miriam BEN-HAROSH, Yves BERTRAND, Trudy BUITENKAMP, Kenneth CALDWELL, Ricardo DRUT, Ashley V GEERLINKS, Gil GILAD, John GRAINGER, Stephanie HAOUY, Nicholas HEANEY, Mary HUANG, Danielle INGHAM, Zdenka KŘENOVÁ, Michaela KUHLEN, Thomas LEHRNBECHER, Atsushi MANABE, Felix NIGGLI, Claudia PARIS, Shoshana REVEL-VILK, Pierre ROHRLICH, Mohamad G SINNO, Tomasz SZCZEPANSKI, Melanie TAMESBERGER, Rajasekharan WARRIER, Matthias WOLFL, Ronit NIREL, Shai IZRAELI, Arndt BORKHARDT and Kjeld SCHMIEGELOW. EBV-driven lymphoid neoplasms associated with pediatric ALL maintenance therapy. \textit{Blood}. Washington DC, USA: American Society of Hematology, 2023, vol.~141, No~7, p.~743-755. ISSN~0006-4971. Available from: https://dx.doi.org/10.1182/blood.2022016975.

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