A novel founder MSH2 deletion in Ethiopian Jews is mainly associated with early-onset colorectal cancer

Fam Cancer. 2022 Apr;21(2):181-188. doi: 10.1007/s10689-021-00249-x. Epub 2021 Apr 10.

Abstract

Lynch syndrome is an inherited cancer predisposition syndrome caused by germline defects in any of the mismatch repair (MMR) genes. Diagnosis of carriers makes precision prevention, early detection, and tailored treatment possible. Herein we report a novel founder deletion of 18,758 bp, mediated by Alu repeats on both sides, detected in Ethiopian Jews. The deletion, which encompasses exon 9-10 of the MSH2 coding sequence, is associated mainly with early-onset MSH2/MSH6-deficient colorectal cancer (CRC) and liposarcoma. Testing of 35 members of 5 seemingly unrelated families of Ethiopian origin yielded 10/21 (48%) carriers, of whom 9 had CRC. Age at first tumor diagnosis ranged from 16 to 89 years. Carriers from the oldest generations were diagnosed after age 45 years (mean 57), and carriers from the younger generation were diagnosed before age 45 years (mean 30). Awareness of this founder deletion is important to improve patient diagnosis, institute surveillance from an early age, and refer patients for genetic counseling addressing the risk of bi-allelic constitutional MMR deficiency syndrome.

Keywords: Anticipation; Deletion; Founder mutation; Lynch syndrome; MSH2.

Publication types

  • Research Support, Non-U.S. Gov't

MeSH terms

  • Adolescent
  • Adult
  • Aged
  • Aged, 80 and over
  • Colorectal Neoplasms* / genetics
  • Colorectal Neoplasms, Hereditary Nonpolyposis* / diagnosis
  • DNA Mismatch Repair / genetics
  • Ethiopia
  • Germ-Line Mutation
  • Humans
  • Jews / genetics
  • Middle Aged
  • MutS Homolog 2 Protein / genetics
  • Young Adult

Substances

  • MSH2 protein, human
  • MutS Homolog 2 Protein