Identifying gene disruptions in novel balanced de novo constitutional translocations in childhood cancer patients by whole-genome sequencing

Genet Med. 2015 Oct;17(10):831-5. doi: 10.1038/gim.2014.189. Epub 2015 Jan 8.

Abstract

Purpose: We applied whole-genome sequencing (WGS) to children diagnosed with neoplasms and found to carry apparently balanced constitutional translocations to discover novel genic disruptions.

Methods: We applied the structural variation (SV) calling programs CREST, BreakDancer, SV-STAT, and CGAP-CNV, and we developed an annotative filtering strategy to achieve nucleotide resolution at the translocations.

Results: We identified the breakpoints for t(6;12)(p21.1;q24.31), disrupting HNF1A in a patient diagnosed with hepatic adenomas and maturity-onset diabetes of the young (MODY). Translocation as the disruptive event of HNF1A, a gene known to be involved in MODY3, has not been previously reported. In a subject with Hodgkin lymphoma and subsequent low-grade glioma, we identified t(5;18)(q35.1;q21.2), disrupting both SLIT3 and DCC, genes previously implicated in both glioma and lymphoma.

Conclusion: These examples suggest that implementing clinical WGS in the diagnostic workup of patients with novel but apparently balanced translocations may reveal unanticipated disruption of disease-associated genes and aid in prediction of the clinical phenotype.

Publication types

  • Research Support, N.I.H., Extramural

MeSH terms

  • Adenoma / diagnosis
  • Adenoma / genetics
  • Adult
  • Age Factors
  • Base Sequence
  • Child
  • Child, Preschool
  • Chromosome Breakpoints
  • Chromosomes, Human, Pair 12
  • Chromosomes, Human, Pair 18
  • Chromosomes, Human, Pair 5
  • Chromosomes, Human, Pair 6
  • DCC Receptor
  • DNA Copy Number Variations
  • Diabetes Mellitus, Type 2 / diagnosis
  • Diabetes Mellitus, Type 2 / genetics
  • Female
  • Genetic Association Studies*
  • Genome, Human*
  • Genomics
  • High-Throughput Nucleotide Sequencing*
  • Humans
  • INDEL Mutation
  • Liver Neoplasms / diagnosis
  • Liver Neoplasms / genetics
  • Male
  • Membrane Proteins / genetics
  • Molecular Sequence Data
  • Neoplasms / diagnosis
  • Neoplasms / genetics*
  • Polymorphism, Single Nucleotide
  • Receptors, Cell Surface / genetics
  • Translocation, Genetic*
  • Tumor Suppressor Proteins / genetics

Substances

  • DCC Receptor
  • DCC protein, human
  • Membrane Proteins
  • Receptors, Cell Surface
  • SLIT3 protein, human
  • Tumor Suppressor Proteins

Supplementary concepts

  • Hepatic Adenomas, Familial
  • Maturity-Onset Diabetes of the Young, Type 3