International cooperative study identifies treatment strategy in childhood ambiguous lineage leukemia
Publikation: Bidrag til tidsskrift › Tidsskriftartikel › Forskning › fagfællebedømt
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International cooperative study identifies treatment strategy in childhood ambiguous lineage leukemia. / Hrusak, Ondrej; de Haas, Valerie; Stancikova, Jitka; Vakrmanova, Barbora; Janotova, Iveta; Mejstrikova, Ester; Capek, Vaclav; Trka, Jan; Zaliova, Marketa; Luks, Ales; Bleckmann, Kirsten; Möricke, Anja; Irving, Julie; Konatkowska, Benigna; Alexander, Thomas B; Inaba, Hiroto; Schmiegelow, Kjeld; Stokley, Simone; Zemanova, Zuzana; Moorman, Anthony V; Rossi, Jorge Gabriel; Felice, Maria Sara; Dalla-Pozza, Luciano; Morales, Jessa; Dworzak, Michael; Buldini, Barbara; Basso, Giuseppe; Campbell, Myriam; Cabrera, Maria Elena; Marinov, Neda; Elitzur, Sarah; Izraeli, Shai; Luria, Drorit; Feuerstein, Tamar; Kolenova, Alexandra; Svec, Peter; Kreminska, Olena; Rabin, Karen R; Polychronopoulou, Sophia; da Costa, Elaine; Marquart, Hanne Vibeke; Kattamis, Antonis; Ratei, Richard; Reinhardt, Dirk; Choi, John K; Schrappe, Martin; Stary, Jan.
I: Blood, Bind 132, Nr. 3, 2018, s. 264-276.Publikation: Bidrag til tidsskrift › Tidsskriftartikel › Forskning › fagfællebedømt
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T1 - International cooperative study identifies treatment strategy in childhood ambiguous lineage leukemia
AU - Hrusak, Ondrej
AU - de Haas, Valerie
AU - Stancikova, Jitka
AU - Vakrmanova, Barbora
AU - Janotova, Iveta
AU - Mejstrikova, Ester
AU - Capek, Vaclav
AU - Trka, Jan
AU - Zaliova, Marketa
AU - Luks, Ales
AU - Bleckmann, Kirsten
AU - Möricke, Anja
AU - Irving, Julie
AU - Konatkowska, Benigna
AU - Alexander, Thomas B
AU - Inaba, Hiroto
AU - Schmiegelow, Kjeld
AU - Stokley, Simone
AU - Zemanova, Zuzana
AU - Moorman, Anthony V
AU - Rossi, Jorge Gabriel
AU - Felice, Maria Sara
AU - Dalla-Pozza, Luciano
AU - Morales, Jessa
AU - Dworzak, Michael
AU - Buldini, Barbara
AU - Basso, Giuseppe
AU - Campbell, Myriam
AU - Cabrera, Maria Elena
AU - Marinov, Neda
AU - Elitzur, Sarah
AU - Izraeli, Shai
AU - Luria, Drorit
AU - Feuerstein, Tamar
AU - Kolenova, Alexandra
AU - Svec, Peter
AU - Kreminska, Olena
AU - Rabin, Karen R
AU - Polychronopoulou, Sophia
AU - da Costa, Elaine
AU - Marquart, Hanne Vibeke
AU - Kattamis, Antonis
AU - Ratei, Richard
AU - Reinhardt, Dirk
AU - Choi, John K
AU - Schrappe, Martin
AU - Stary, Jan
N1 - © 2018 by The American Society of Hematology.
PY - 2018
Y1 - 2018
N2 - Despite attempts to improve the definitions of ambiguous lineage leukemia (ALAL) during the last 2 decades, general therapy recommendations are missing. Herein, we report a large cohort of children with ALAL and propose a treatment strategy. A retrospective multinational study (International Berlin-Frankfurt-Münster Study of Leukemias of Ambiguous Lineage [iBFM-AMBI2012]) of 233 cases of pediatric ALAL patients is presented. Survival statistics were used to compare the prognosis of subsets and types of treatment. Five-year event-free survival (EFS) of patients with acute lymphoblastic leukemia (ALL)-type primary therapy (80% ± 4%) was superior to that of children who received acute myeloid leukemia (AML)-type or combined-type treatment (36% ± 7.2% and 50% ± 12%, respectively). When ALL- or AML-specific gene fusions were excluded, 5-year EFS of CD19+ leukemia was 83% ± 5.3% on ALL-type primary treatment compared with 0% ± 0% and 28% ± 14% on AML-type and combined-type primary treatment, respectively. Superiority of ALL-type treatment was documented in single-population mixed phenotype ALAL (using World Health Organization and/or European Group for Immunophenotyping of Leukemia definitions) and bilineal ALAL. Treatment with ALL-type protocols is recommended for the majority of pediatric patients with ALAL, including cases with CD19+ ALAL. AML-type treatment is preferred in a minority of ALAL cases with CD19- and no other lymphoid features. No overall benefit of transplantation was documented, and it could be introduced in some patients with a poor response to treatment. As no clear indicator was found for a change in treatment type, this is to be considered only in cases with ≥5% blasts after remission induction. The results provide a basis for a prospective trial.
AB - Despite attempts to improve the definitions of ambiguous lineage leukemia (ALAL) during the last 2 decades, general therapy recommendations are missing. Herein, we report a large cohort of children with ALAL and propose a treatment strategy. A retrospective multinational study (International Berlin-Frankfurt-Münster Study of Leukemias of Ambiguous Lineage [iBFM-AMBI2012]) of 233 cases of pediatric ALAL patients is presented. Survival statistics were used to compare the prognosis of subsets and types of treatment. Five-year event-free survival (EFS) of patients with acute lymphoblastic leukemia (ALL)-type primary therapy (80% ± 4%) was superior to that of children who received acute myeloid leukemia (AML)-type or combined-type treatment (36% ± 7.2% and 50% ± 12%, respectively). When ALL- or AML-specific gene fusions were excluded, 5-year EFS of CD19+ leukemia was 83% ± 5.3% on ALL-type primary treatment compared with 0% ± 0% and 28% ± 14% on AML-type and combined-type primary treatment, respectively. Superiority of ALL-type treatment was documented in single-population mixed phenotype ALAL (using World Health Organization and/or European Group for Immunophenotyping of Leukemia definitions) and bilineal ALAL. Treatment with ALL-type protocols is recommended for the majority of pediatric patients with ALAL, including cases with CD19+ ALAL. AML-type treatment is preferred in a minority of ALAL cases with CD19- and no other lymphoid features. No overall benefit of transplantation was documented, and it could be introduced in some patients with a poor response to treatment. As no clear indicator was found for a change in treatment type, this is to be considered only in cases with ≥5% blasts after remission induction. The results provide a basis for a prospective trial.
U2 - 10.1182/blood-2017-12-821363
DO - 10.1182/blood-2017-12-821363
M3 - Journal article
C2 - 29720486
VL - 132
SP - 264
EP - 276
JO - Blood
JF - Blood
SN - 0006-4971
IS - 3
ER -
ID: 222319471