Author(s): Case MC, Matheson E, Minto L, Hassan R, Harrison CJ, Bown NP, Bailey S, Vormoor J, Hall AG, Irving JAE
Abstract: Deregulation of the RAS-RAF-mitogen-activated protein kinase/extracellular signal-regulated kinase (ERK) kinase (MEK)-ERK signaling cascade is often caused by somatic mutations in genes encoding proteins which influence the activity of this pathway and include NRAS, KRAS2, FLT3, PTPN11, and BRAF. We report the first comprehensive mutational screen of key exons of these genes in a large cohort of unselected acute lymphoblastic leukemia (ALL) cases at diagnosis (n = 86) and in a more selected cohort at disease recurrence (n = 47) using the sensitive method of denaturing high-performance liquid chromatography. We show that somatic mutations that deregulate the pathway constitute one of the most common genetic aberrations in childhood ALL (cALL), being found in 35% of diagnostic and 25% of relapse samples. In matched presentation/relapse pairs, mutations predominating at relapse could be shown to be present at very low levels at diagnosis using allele-specific PCR, thus implicating the mutated clone in disease progression. Importantly, in primary samples, we show that mutations are associated with activated ERK and differential cytotoxicity to MEK-ERK inhibitors was shown for some patients. Inhibitors of the pathway, which are currently undergoing clinical trial, may be a novel therapeutic option for cALL, particularly at relapse.
Keywords: Adolescent Blotting, Western Cell Survival Child Child, Preschool Cohort Studies DNA Mutational Analysis Enzyme Inhibitors/pharmacology Exons/genetics Extracellular Signal-Regulated MAP Kinases/antagonists & inhibitors/metabolism Female Genes, ras/*physiology Humans In Situ Hybridization, Fluorescence Infant Male Mitogen-Activated Protein Kinase Kinases/antagonists & inhibitors/metabolism Mitogen-Activated Protein Kinases/antagonists & inhibitors/metabolism Mutation/*genetics Neoplasm Recurrence, Local/*genetics/pathology Peptide Fragments Ploidies Precursor Cell Lymphoblastic Leukemia-Lymphoma/*genetics/pathology Protein Tyrosine Phosphatase, Non-Receptor Type 11/genetics Proto-Oncogene Proteins/*genetics Proto-Oncogene Proteins B-raf/genetics Remission Induction *Signal Transduction Tumor Cells, Cultured fms-Like Tyrosine Kinase 3/genetics ras Proteins/*genetics
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Professor Andy Hall
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Dr Julie Irving
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