Fusion of the Lumican (LUM) Gene With the Ubiquitin Specific Peptidase 6 (USP6) Gene in an Aneurysmal Bone Cyst Carrying a t(12;17)(q21;p13) Chromosome Translocation

Cancer Genomics Proteomics. 2020 Sep-Oct;17(5):555-561. doi: 10.21873/cgp.20211.

Abstract

Background/aim: Aneurysmal bone cyst is a benign bone lesion with a strong tendency to recur. The rearrangement of chromosome band 17p13/USP6 gene is now considered a characteristic genetic feature of aneurysmal bone cyst, with t(16;17)(q22;p13)/CDH11-USP6 as the most frequent chromosomal aberration/fusion gene. We report a novel variant translocation leading to a new fusion gene in an aneurysmal bone cyst.

Materials and methods: Genetic analyses were performed on an aneurysmal bone cyst found in the tibia of a child.

Results: G-banding chromosome analysis yielded the karyotype 46,XX,t(12;17)(q21;p13)[5]/46,XX[2]. FISH analysis with a USP6 break-apart probe showed rearrangement of USP6. RNA sequencing detected LUM-USP6 and USP6-LUM fusion transcripts which were subsequently verified by RT-PCR/Sanger sequencing. The two genes exchanged 5'- non-coding exons. Thus, promoter swapping between USP6 and LUM had taken place.

Conclusion: We report a novel t(12;17)(q21;p13) chromosome translocation which gave rise to a LUM-USP6 fusion in an aneurysmal bone cyst.

Keywords: Aneurysmal bone cyst; LUM; LUM-USP6 fusion gene; USP6; chromosome translocation.

Publication types

  • Case Reports

MeSH terms

  • Bone Cysts, Aneurysmal / diagnosis
  • Bone Cysts, Aneurysmal / genetics*
  • Bone Cysts, Aneurysmal / pathology
  • Child
  • Chromosomes, Human, Pair 16 / genetics
  • Chromosomes, Human, Pair 17 / genetics
  • Female
  • Gene Fusion
  • Humans
  • In Situ Hybridization, Fluorescence
  • Karyotyping
  • Lumican / genetics*
  • Promoter Regions, Genetic / genetics
  • Sequence Analysis, RNA
  • Translocation, Genetic*
  • Ubiquitin Thiolesterase / genetics*

Substances

  • LUM protein, human
  • Lumican
  • USP6 protein, human
  • Ubiquitin Thiolesterase