Clonal evidence for the development of neuroblastoma with extensive copy-neutral loss of heterozygosity arising in a mature teratoma

Cancer Sci. 2021 Jul;112(7):2921-2927. doi: 10.1111/cas.14931. Epub 2021 May 6.

Abstract

Mature teratomas are usually benign tumors that rarely undergo malignant transformation. We report an advanced neuroblastoma arising in a mature teratoma of the ovary. Whole-exome sequencing identified extensive copy-neutral loss of heterozygosity (LOH) in both neuroblastoma and teratoma elements, suggesting that the neuroblastoma evolved from the teratoma. In addition, several truncating germline heterozygous variants in tumor suppressor genes, including RBL2 and FBXW12, became homozygous as a result of LOH. Collectively, we speculate that extensive LOH in teratoma cells may force heterozygous germline variants to become homozygous, which, in turn, may contribute to the development of neuroblastoma with the acquisition of additional chromosomal changes.

Keywords: cancer genome/genetics; copy-neutral loss of heterozygosity; genomic analysis; malignant transformation; neuroblastoma; teratoma.

Publication types

  • Case Reports

MeSH terms

  • Adolescent
  • Exome Sequencing
  • F-Box Proteins / genetics
  • Female
  • Germ-Line Mutation*
  • Homozygote
  • Humans
  • Loss of Heterozygosity*
  • Neoplasms, Multiple Primary / drug therapy
  • Neoplasms, Multiple Primary / genetics*
  • Neoplasms, Multiple Primary / pathology
  • Neuroblastoma / drug therapy
  • Neuroblastoma / genetics*
  • Neuroblastoma / pathology
  • Ovarian Neoplasms / drug therapy
  • Ovarian Neoplasms / genetics*
  • Ovarian Neoplasms / pathology
  • Retinoblastoma-Like Protein p130 / genetics
  • Teratoma / drug therapy
  • Teratoma / genetics*
  • Teratoma / pathology

Substances

  • F-Box Proteins
  • FBXW12 protein, human
  • RBL2 protein, human
  • Retinoblastoma-Like Protein p130