Case Report: A Low-grade Uterine Leiomyosarcoma Showing Multiple Genetic Aberrations Including a Bi-allelic Loss of the Retinoblastoma Gene Locus, as well as Germ-line Uniparental Disomy for Part of the Long Arm of Chromosome 22

Anticancer Res. 2017 May;37(5):2233-2237. doi: 10.21873/anticanres.11559.

Abstract

Background: Uterine leiomyosarcomas are rare tumors with adverse prognosis. Recently, it has been suggested that a possible genetic subgroup of these tumors might be characterized by bi-allelic deletions of the RB1 locus. Here we report another uterine leiomyosarcoma with bi-allelic deletion of RB1 along with other genetic alterations.

Case report: A 52-year-old patient was admitted to the hospital for surgical removal of a polyp-like lesion in the uterine cavity. Histological examination revealed a grade 1 leiomyosarcoma with atypical mitoses and areas corresponding to a leiomyoma with bizarre nuclei.

Results and conclusion: This is the third case of a uterine leiomyosarcoma revealing bi-allelic RB1 deletions. Thus, in the absence of monosomy 14 and/or mutations of MED12, this genetic alteration seems, indeed, to constitute a separate entity of these tumors. Histological analysis of the tumor along with its genetic intratumoral heterogeneity suggests its origin to be from a leiomyoma with bizarre nuclei. Furthermore, of considerable interest in the case presented here, is the identification of a large segment of chromosome 22 showing uniparental disomy. Along with the case presented here, recent data show that a genetic classification of all uterine leiomyosarcomas is recommended to reveal more information about clinical correlations of their different genetic subtypes. Due to array-based methods these analyses can be well-carried out using paraffin-embedded samples.

Keywords: LOH; RB1; UPD; Uterine leiomyosarcoma; deletion; genetic alterations; loss of heterozygosity; uniparental disomy.

Publication types

  • Case Reports

MeSH terms

  • Chromosomes, Human, Pair 22
  • Female
  • Genetic Loci
  • Humans
  • Leiomyosarcoma / genetics*
  • Loss of Heterozygosity
  • Middle Aged
  • Retinoblastoma Binding Proteins / genetics
  • Tumor Suppressor Protein p53 / genetics
  • Ubiquitin-Protein Ligases / genetics
  • Uniparental Disomy
  • Uterine Neoplasms / genetics*

Substances

  • RB1 protein, human
  • Retinoblastoma Binding Proteins
  • TP53 protein, human
  • Tumor Suppressor Protein p53
  • Ubiquitin-Protein Ligases