Microarray-based copy number analysis of neurofibromatosis type-1 (NF1)-associated malignant peripheral nerve sheath tumors reveals a role for Rho-GTPase pathway genes in NF1 tumorigenesis

Hum Mutat. 2012 Apr;33(4):763-76. doi: 10.1002/humu.22044. Epub 2012 Mar 5.

Abstract

Neurofibromatosis type-1 (NF1) is associated with the growth of benign and malignant tumors. Approximately 15% of NF1 patients develop malignant peripheral nerve sheath tumors (MPNSTs), underlining the need to identify specific diagnostic/prognostic biomarkers associated with MPNST development. The Affymetrix Genome-Wide Human single-nucleotide polymorphism (SNP) Array 6.0 was used to perform SNP genotyping and copy number alteration (CNA), loss-of-heterozygosity (LOH), and copy number neutral-LOH (CNN-LOH) analyses of DNA isolated from 15 MPNSTs, five benign plexiform neurofibromas (PNFs), and patient-matched lymphocyte DNAs. MPNSTs exhibited high-level CNN-LOH, with recurrent changes occurring in MPNSTs but not PNFs. CNN-LOH was evident in MPNSTs but occurred less frequently than genomic deletions. CNAs involving the ITGB8, PDGFA, Ras-related C3 botulinum toxin substrate 1 (RAC1) (7p21-p22), PDGFRL (8p22-p21.3), and matrix metallopeptidase 12 (MMP12) (11q22.3) genes were specific to MPNSTs. Pathway analysis revealed the MPNST-specific amplification of seven Rho-GTPase pathway genes and several cytoskeletal remodeling/cell adhesion genes. In knockdown experiments employing short-hairpin RAC1, ROCK2, PTK2, and LIMK1 RNAs to transfect both control and MPNST-derived cell lines, cell adhesion was significantly increased in the MPNST cell lines, whereas wound healing, cell migration, and invasiveness were reduced, consistent with a role for these Rho-GTPase pathway genes in MPNST development and metastasis. These results suggest new targets for therapeutic intervention in relation to MPNSTs.

Publication types

  • Research Support, Non-U.S. Gov't

MeSH terms

  • Cell Adhesion / genetics
  • Cell Movement / genetics
  • Focal Adhesion Kinase 1 / genetics
  • GTP Phosphohydrolases / genetics
  • GTP Phosphohydrolases / metabolism*
  • Gene Knockdown Techniques
  • Humans
  • Lim Kinases / genetics
  • Loss of Heterozygosity*
  • Matrix Metalloproteinase 12 / genetics
  • Nerve Sheath Neoplasms / genetics*
  • Nerve Sheath Neoplasms / pathology
  • Neurofibromatosis 1 / genetics*
  • Neurofibromatosis 1 / pathology
  • Oligonucleotide Array Sequence Analysis
  • Platelet-Derived Growth Factor / genetics
  • Polymorphism, Single Nucleotide
  • Receptors, Platelet-Derived Growth Factor / genetics
  • Tumor Suppressor Proteins / genetics
  • rac1 GTP-Binding Protein / genetics
  • rho-Associated Kinases / genetics

Substances

  • PDGFRL protein, human
  • Platelet-Derived Growth Factor
  • RAC1 protein, human
  • Tumor Suppressor Proteins
  • platelet-derived growth factor A
  • Receptors, Platelet-Derived Growth Factor
  • Focal Adhesion Kinase 1
  • PTK2 protein, human
  • LIMK1 protein, human
  • Lim Kinases
  • ROCK2 protein, human
  • rho-Associated Kinases
  • Matrix Metalloproteinase 12
  • GTP Phosphohydrolases
  • rac1 GTP-Binding Protein