A novel germline MLH1 mutation causing Lynch Syndrome in patients from the Republic of Macedonia

Croat Med J. 2012 Oct;53(5):496-501. doi: 10.3325/cmj.2012.53.496.

Abstract

Aim: To implement molecular analysis in the clinical diagnosis and management of Lynch syndrome (LS).

Methods: We analyzed the mutations in MLH1 and MSH2 in the selected LS families from the Republic of Macedonia.

Results: We performed the very first genetic identification of LS families and characterized a novel mutation. The novel nonsense germline point mutation c.392C>G in the codon 131 of MLH1(S131X) was identified as the underlying genetic cause of LS in three families. The haplotype analysis suggested a founder effect of this mutation in our population.

Conclusion: We expect to detect the mutation in other LS patients from the region, and recommend cost-effective screening for this mutation by restriction fragment length polymorphism-polymerase chain reaction or DNA sequencing of MLH1 Exon5 prior to full genetic testing in all LS suspects of Macedonian ancestry.

Publication types

  • Research Support, Non-U.S. Gov't

MeSH terms

  • Adaptor Proteins, Signal Transducing / genetics*
  • Base Sequence
  • Colorectal Neoplasms, Hereditary Nonpolyposis / genetics*
  • DNA Mutational Analysis
  • Female
  • Founder Effect
  • Germ-Line Mutation*
  • Haplotypes
  • Humans
  • Male
  • Middle Aged
  • Multiplex Polymerase Chain Reaction
  • MutL Protein Homolog 1
  • MutS Homolog 2 Protein / genetics
  • Mutation, Missense*
  • Nuclear Proteins / genetics*
  • Pedigree
  • Point Mutation*
  • Prospective Studies
  • Republic of North Macedonia
  • Sequence Analysis, DNA

Substances

  • Adaptor Proteins, Signal Transducing
  • MLH1 protein, human
  • Nuclear Proteins
  • MSH2 protein, human
  • MutL Protein Homolog 1
  • MutS Homolog 2 Protein