Chromosomal imbalances detected by comparative genomic hybridisation in atypical teratoid/rhabdoid tumours

Childs Nerv Syst. 2004 Apr;20(4):221-4. doi: 10.1007/s00381-003-0909-8. Epub 2004 Feb 4.

Abstract

Introduction: Atypical teratoid/rhabdoid tumours (AT/RT) are highly malignant embryonal tumours of the brain composed of rhabdoid cells. Inactivating mutations of the hSNF5/INI-1 gene located in the chromosomal region 22q11.2 are regarded as a crucial step in their molecular pathogenesis. Apart from monosomy or deletions of chromosome 22 not much data exists on additional chromosomal aberrations.

Methods: We investigated seven primary AT/RT by comparative genomic hybridisation (CGH) and found DNA copy number changes in each case.

Results: These consisted of loss of 22q in 7 out of 7 (100%) and loss of 19 in 3 out of 7 (43%) patients. In 4/7 AT/RT (57%), loss of chromosome 22q was the sole aberration whereas one patient showed additional losses of 16p, 17p and 20q.

Conclusions: Our CGH data suggest that apart from monosomy 22 additional genetic pathways may seem feasible for a subset of AT/RT that is yet to be defined. Furthermore, this study also emphasises the potential practical value of loss of chromosome 22 as a diagnostic marker for AT/RT.

Publication types

  • Comparative Study

MeSH terms

  • Brain Neoplasms / genetics*
  • Child, Preschool
  • Chromosomal Proteins, Non-Histone
  • Chromosome Aberrations*
  • Chromosomes, Human, Pair 22*
  • DNA-Binding Proteins / genetics
  • Female
  • Humans
  • Infant
  • Infant, Newborn
  • Male
  • Mutation
  • Nucleic Acid Hybridization
  • Rhabdoid Tumor / genetics*
  • SMARCB1 Protein
  • Transcription Factors

Substances

  • Chromosomal Proteins, Non-Histone
  • DNA-Binding Proteins
  • SMARCB1 Protein
  • SMARCB1 protein, human
  • Transcription Factors