International cooperative study identifies treatment strategy in childhood ambiguous lineage leukemia
Language English Country United States Media print-electronic
Document type Journal Article, Multicenter Study, Research Support, Non-U.S. Gov't
PubMed
29720486
DOI
10.1182/blood-2017-12-821363
PII: S0006-4971(20)32060-7
Knihovny.cz E-resources
- MeSH
- Leukemia, Biphenotypic, Acute diagnosis etiology therapy MeSH
- Biomarkers MeSH
- Child MeSH
- Infant MeSH
- Combined Modality Therapy MeSH
- Humans MeSH
- Disease Management MeSH
- Adolescent MeSH
- Disease Susceptibility MeSH
- Biomarkers, Tumor MeSH
- Infant, Newborn MeSH
- Child, Preschool MeSH
- Prognosis MeSH
- Proportional Hazards Models MeSH
- Treatment Outcome MeSH
- Check Tag
- Child MeSH
- Infant MeSH
- Humans MeSH
- Adolescent MeSH
- Male MeSH
- Infant, Newborn MeSH
- Child, Preschool MeSH
- Female MeSH
- Publication type
- Journal Article MeSH
- Multicenter Study MeSH
- Research Support, Non-U.S. Gov't MeSH
- Names of Substances
- Biomarkers MeSH
- Biomarkers, Tumor MeSH
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.
1st Department of Pediatrics University of Athens Athens Greece
Bioinformatics Centre 2nd Faculty of Medicine Charles University Prague Czech Republic
Childhood Leukemia Investigation Prague Prague Czech Republic
Comenius University Children's Hospital Bratislava Slovakia
Department of Oncology St Jude Children's Research Hospital Memphis TN
Department of Pathology St Jude Children's Research Hospital Memphis TN
Department of Pediatric Hematology Oncology Aghia Sophia Children's Hospital Athens Greece
Department of Pediatrics University Medical Center Schleswig Holstein Campus Kiel Kiel Germany
Dutch Childhood Oncology Group The Hague The Netherlands
Helios Klinikum Berlin Buch Berlin Germany
Hematology and Oncology Hospital Nacional de Pediatría Prof Dr J P Garrahan Buenos Aires Argentina
Hospital del Salvador Universidad de Chile Santiago Chile
Immunology and Rheumatology and
Institute of Medicine Faculty of Medicine University of Copenhagen Copenhagen Denmark
Northern Institute for Cancer Research Newcastle University Newcastle upon Tyne United Kingdom
Nottingham University Hospitals NHS Trust Nottingham United Kingdom
Pediatric Hematology and Oncology University Children's Hospital Essen Essen Germany; and
Pediatric Hematology Oncology Baylor College of Medicine Houston TX
Pediatric Hematology Oncology Schneider Children's Medical Center Petah Tikva Israel
Pediatrics Department Faculty of Medicine Federal University of Rio de Janeiro Rio de Janeiro Brazil
Princess Máxima Center for Pediatric Oncology Utrecht The Netherlands
Sackler Faculty of Medicine Tel Aviv University Tel Aviv Israel
Sydney Children's Hospital Network Westmead NSW Australia
University Medical Center Christian Albrechts University Kiel Kiel Germany
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