A novel integrated cytogenetic and genomic classification refines risk stratification in pediatric acute lymphoblastic leukemia

Blood. 2014 Aug 28;124(9):1434-44. doi: 10.1182/blood-2014-03-562918. Epub 2014 Jun 23.

Abstract

Recent genomic studies have provided a refined genetic map of acute lymphoblastic leukemia (ALL) and increased the number of potential prognostic markers. Therefore, we integrated copy-number alteration data from the 8 most commonly deleted genes, subordinately, with established chromosomal abnormalities to derive a 2-tier genetic classification. The classification was developed using 809 ALL97/99 patients and validated using 742 United Kingdom (UK)ALL2003 patients. Good-risk (GR) genetic features included ETV6-RUNX1, high hyperdiploidy, normal copy-number status for all 8 genes, isolated deletions affecting ETV6/PAX5/BTG1, and ETV6 deletions with a single additional deletion of BTG1/PAX5/CDKN2A/B. All other genetic features were classified as poor risk (PR). Three-quarters of UKALL2003 patients had a GR genetic profile and a significantly improved event-free survival (EFS) (94%) compared with patients with a PR genetic profile (79%). This difference was driven by a lower relapse rate (4% vs 17%), was seen across all patient subgroups, and was independent of other risk factors. Even genetic GR patients with minimal residual disease (>0.01%) at day 29 had an EFS in excess of 90%. In conclusion, the integration of genomic and cytogenetic data defines 2 subgroups with distinct responses to treatment and identifies a large subset of children suitable for treatment deintensification.

Publication types

  • Research Support, Non-U.S. Gov't
  • Validation Study

MeSH terms

  • Adolescent
  • Child
  • Child, Preschool
  • Cohort Studies
  • Core Binding Factor Alpha 2 Subunit / genetics
  • Cytogenetic Analysis
  • ETS Translocation Variant 6 Protein
  • Female
  • Gene Deletion
  • Gene Dosage
  • Genes, p16
  • Genomics
  • Humans
  • Infant
  • Kaplan-Meier Estimate
  • Male
  • Neoplasm Proteins / genetics
  • Oncogene Proteins, Fusion / genetics
  • PAX5 Transcription Factor / genetics
  • Precursor B-Cell Lymphoblastic Leukemia-Lymphoma / classification
  • Precursor B-Cell Lymphoblastic Leukemia-Lymphoma / drug therapy
  • Precursor B-Cell Lymphoblastic Leukemia-Lymphoma / genetics*
  • Prognosis
  • Proto-Oncogene Proteins c-ets / genetics
  • Repressor Proteins / genetics
  • Risk Factors

Substances

  • Core Binding Factor Alpha 2 Subunit
  • Neoplasm Proteins
  • Oncogene Proteins, Fusion
  • PAX5 Transcription Factor
  • PAX5 protein, human
  • Proto-Oncogene Proteins c-ets
  • Repressor Proteins
  • TEL-AML1 fusion protein
  • BTG1 protein, human