Diagnostic yield of multigene panel testing in an Israeli cohort: enrichment of low-penetrance variants

Breast Cancer Res Treat. 2020 Jun;181(2):445-453. doi: 10.1007/s10549-020-05633-2. Epub 2020 Apr 18.

Abstract

Background: Carriers of pathogenic variants (PVs) in moderate-high-penetrance cancer susceptibility genes are offered tailored surveillance schemes for early cancer diagnosis. The clinical implications of low-penetrance variant carriers are less clear.

Methods: Clinical and demographic data were retrieved for a cohort of Israeli individuals who underwent oncogenetic testing by the 30-gene cancer panel at Color Genomics laboratory, between 04/2013 and 12/2018.

Results: Of 758 genotyped individuals, 504 had been diagnosed with cancer prior to testing: 283 (56%) had breast cancer and 106 (21%) colorectal cancer. Pathogenic or likely pathogenic (P/LP) variants were detected in 123 (16%) individuals. Overall, 44 different P/LP variants were detected in 18/30 cancer susceptibility genes; 20 of them were founder/recurrent mutations. Of the carriers, 39 (32%), 10 (8%), and 74 (60%) carried high-, moderate-, or low-penetrance variants, respectively. After excluding low-penetrance variants, 7% (33/504) of all cancer patients, 6% of breast or ovarian cancer patients were found to be carriers, as well as 7% (14/203) of individuals with colonic polyps, and 4% (11/254) of cancer-free individuals.

Conclusions: The diagnostic yield of moderate- and high-penetrance PVs using multigene panel testing was 6%, with 3.7% carriers of non-recurrent PVs. This yield should be discussed during pre-test counseling, and emphasizes the need for harmonized recommendations regarding clinical implications of low-penetrance variants.

Keywords: Cancer predisposition; Clinical utility; Inherited cancer syndromes; Low-penetrance variants; Multi-gene panel testing; Recurrent mutations.

MeSH terms

  • Adolescent
  • Adult
  • Aged
  • Aged, 80 and over
  • Biomarkers, Tumor / genetics*
  • Breast Neoplasms / diagnosis*
  • Breast Neoplasms / epidemiology
  • Breast Neoplasms / genetics
  • Cohort Studies
  • Early Detection of Cancer / methods*
  • Ethnicity / genetics*
  • Ethnicity / statistics & numerical data
  • Female
  • Follow-Up Studies
  • Genetic Predisposition to Disease*
  • Genetic Testing / methods*
  • Humans
  • Israel / epidemiology
  • Middle Aged
  • Mutation*
  • Penetrance
  • Prognosis
  • Young Adult

Substances

  • Biomarkers, Tumor