Characterization of uterine leiomyomas by whole-genome sequencing

N Engl J Med. 2013 Jul 4;369(1):43-53. doi: 10.1056/NEJMoa1302736. Epub 2013 Jun 5.

Abstract

Background: Uterine leiomyomas are benign but affect the health of millions of women. A better understanding of the molecular mechanisms involved may provide clues to the prevention and treatment of these lesions.

Methods: We performed whole-genome sequencing and gene-expression profiling of 38 uterine leiomyomas and the corresponding myometrium from 30 women.

Results: Identical variants observed in some separate tumor nodules suggested that these nodules have a common origin. Complex chromosomal rearrangements resembling chromothripsis were a common feature of leiomyomas. These rearrangements are best explained by a single event of multiple chromosomal breaks and random reassembly. The rearrangements created tissue-specific changes consistent with a role in the initiation of leiomyoma, such as translocations of the HMGA2 and RAD51B loci and aberrations at the COL4A5-COL4A6 locus, and occurred in the presence of normal TP53 alleles. In some cases, separate events had occurred more than once in single tumor-cell lineages.

Conclusions: Chromosome shattering and reassembly resembling chromothripsis (a single genomic event that results in focal losses and rearrangements in multiple genomic regions) is a major cause of chromosomal abnormalities in uterine leiomyomas; we propose that tumorigenesis occurs when tissue-specific tumor-promoting changes are formed through these events. Chromothripsis has previously been associated with aggressive cancer; its common occurrence in leiomyomas suggests that it also has a role in the genesis and progression of benign tumors. We observed that multiple separate tumors could be seeded from a single lineage of uterine leiomyoma cells. (Funded by the Academy of Finland Center of Excellence program and others.).

Publication types

  • Research Support, Non-U.S. Gov't

MeSH terms

  • Chromosome Aberrations*
  • Chromosome Breakage
  • Chromosome Deletion
  • Collagen Type IV / genetics
  • Female
  • Fumarate Hydratase / deficiency*
  • Fumarate Hydratase / genetics
  • Gene Expression Profiling
  • Gene Rearrangement
  • Genome-Wide Association Study
  • Humans
  • Leiomyoma / genetics*
  • Mediator Complex / genetics*
  • Mutation
  • Myometrium / chemistry
  • Up-Regulation
  • Uterine Neoplasms / genetics*

Substances

  • COL4A5 protein, human
  • COL4A6 protein, human
  • Collagen Type IV
  • MED12 protein, human
  • Mediator Complex
  • Fumarate Hydratase