Involvement of germline DDX1-MYCN duplication in inherited nephroblastoma

Eur J Med Genet. 2013 Dec;56(12):643-7. doi: 10.1016/j.ejmg.2013.10.004. Epub 2013 Oct 24.

Abstract

This report concerns a 3-year-old girl with prenatal bilateral nephroblastomatosis and a family history of nephroblastoma. This girl had a chromosome 8 pericentric inversion inherited from her father. This inversion was observed in healthy individuals of the family and was absent in other individuals suffering from embryonic kidney tumor. We then supposed that another genetic anomaly predisposed her to tumorogenesis. Additional cryptic imbalances are reported in cases of apparently balanced chromosomal rearrangements with an abnormal phenotype. Array-CGH analysis showed a 569 kb duplication at 2p24.3 including the DDX1 and MYCN genes. This duplication was inherited from the patient's father who also had a nephroblastoma. A link between germline MYCN duplication and the occurrence of other embryonic cancers such as neuroblastoma has already been described. We supposed that germline DDX1-MYCN duplication could also be involved in the apparition of nephroblastomas.

Keywords: 2p24.3 duplication; DDX1; MYCN; Nephroblastomatosis; Wilms tumor.

Publication types

  • Case Reports

MeSH terms

  • Adult
  • Carcinogenesis / genetics
  • Child, Preschool
  • Chromosomes, Human, Pair 8 / genetics
  • Comparative Genomic Hybridization
  • DEAD-box RNA Helicases / genetics*
  • Female
  • Gene Duplication*
  • Genetic Predisposition to Disease
  • Germ-Line Mutation*
  • Humans
  • Kidney Neoplasms / diagnosis
  • Kidney Neoplasms / genetics*
  • Male
  • N-Myc Proto-Oncogene Protein
  • Nuclear Proteins / genetics*
  • Oncogene Proteins / genetics*
  • Pedigree
  • Wilms Tumor / diagnosis
  • Wilms Tumor / genetics*

Substances

  • MYCN protein, human
  • N-Myc Proto-Oncogene Protein
  • Nuclear Proteins
  • Oncogene Proteins
  • DDX1 protein, human
  • DEAD-box RNA Helicases