Lynch syndrome screening in patients with young-onset extra-colorectal Lynch syndrome-associated cancers

Int J Clin Oncol. 2024 Nov;29(11):1696-1703. doi: 10.1007/s10147-024-02609-w. Epub 2024 Aug 26.

Abstract

Background: Lynch syndrome (LS) is a hereditary cancer syndrome caused by pathogenic germline variants in mismatch repair (MMR) genes, which predisposes to various types of cancers showing deficient MMR (dMMR). Identification of LS probands is crucial to reduce cancer-related deaths in affected families. Although universal screening is recommended for colorectal and endometrial cancers, and age-restricted screening is proposed as an alternative, LS screening covering a broader spectrum of cancer types is needed. In the current study, we elucidated the rate of dMMR tumors and evaluated the outcome of LS screening in young-onset extra-colorectal LS-associated cancers.

Methods: Immunohistochemistry for MMR proteins were retrospectively performed in a total of 309 tissue samples of endometrial, non-mucinous ovarian, gastric, urothelial, pancreatic, biliary tract, and adrenal cancers in patients < 50 years of age. Clinicopathological information and the results of genetic testing were obtained from medical charts.

Results: There were 24 dMMR tumors (7.8%) including 18 endometrial, three ovarian, two urothelial, and one gastric cancer. Co-occurrence of colorectal cancer and family history of LS-associated cancers was significantly enriched in patients with dMMR tumors. Among the 16 patients with dMMR tumors who were informed of the immunohistochemistry results, five with endometrial and one with urothelial cancer were diagnosed as LS with positive pathogenic variants in MMR genes.

Conclusions: We report the outcome of immunohistochemistry for MMR proteins performed in multiple types of young-onset extra-colorectal LS-associated cancers. Our study demonstrates the feasibility of a comprehensive LS screening program incorporating young-onset patients with various types of extra-colorectal LS-associated cancers.

Keywords: Deficient mismatch repair; Lynch syndrome; Lynch syndrome-associated cancer; Screening; Young-onset.

MeSH terms

  • Adult
  • Age of Onset
  • Colorectal Neoplasms, Hereditary Nonpolyposis* / complications
  • Colorectal Neoplasms, Hereditary Nonpolyposis* / diagnosis
  • Colorectal Neoplasms, Hereditary Nonpolyposis* / genetics
  • DNA Mismatch Repair* / genetics
  • Early Detection of Cancer / methods
  • Female
  • Genetic Testing
  • Humans
  • Male
  • Middle Aged
  • MutL Protein Homolog 1 / genetics
  • MutS Homolog 2 Protein / genetics
  • Retrospective Studies
  • Young Adult

Substances

  • MutL Protein Homolog 1
  • MLH1 protein, human
  • MutS Homolog 2 Protein