Characterization of a novel germline BRCA1 splice variant, c.5332+4delA

Breast Cancer Res Treat. 2018 Apr;168(2):543-550. doi: 10.1007/s10549-017-4595-8. Epub 2017 Nov 28.

Abstract

Purpose: Germline mutations in BRCA1 and BRCA2 confer a significant increase in risk for cancer, and determining pathogenicity of a BRCA variant can guide the clinical management of the disease. About 1/3 of BRCA1 variants reported in the public databases have uncertain clinical significance due to lack of conclusive evidence. This study aims to characterize a novel BRCA1 deletion affecting the + 4 splice donor site identified in an individual with early-onset breast cancer.

Methods: The effect of BRCA1 c.5332+4delA variant on RNA splicing was evaluated by amplifying regions of BRCA1 from cDNA derived from the patient. The proportion of abnormal transcript in the total transcripts was quantified. Loss of heterozygosity (LOH) in tumor tissue was investigated using Sanger sequencing and fragment analysis.

Results: BRCA1 c.5332+4delA caused skipping of exon 21 in patient-derived samples. Semi-quantitative analysis indicated that this aberrant RT-PCR product accounts for about 40% of the total transcript levels. Loss of heterozygosity (LOH) was observed in patient's tumor tissue.

Conclusions: Our results indicate that the BRCA1 c.5332+4delA variant contributes to cancer predisposition through disruption of normal mRNA splicing. We classify this variant as likely pathogenic.

Keywords: BRCA1; Breast cancer; Exon skipping; Germline variant; Loss of heterozygosity; Pathogenic; Splicing; c.5332+4delA.

Publication types

  • Case Reports

MeSH terms

  • Adult
  • Age of Onset
  • BRCA1 Protein / genetics*
  • Base Sequence / genetics
  • Breast Neoplasms / genetics*
  • Female
  • Genetic Predisposition to Disease*
  • Humans
  • Loss of Heterozygosity / genetics
  • Pedigree
  • RNA Splice Sites / genetics*
  • RNA Splicing / genetics*
  • Sequence Deletion

Substances

  • BRCA1 Protein
  • BRCA1 protein, human
  • RNA Splice Sites