Literature DB >> 35386111

Familial Lynch syndrome with early age of onset and confirmed splice site mutation in MSH2: A case report.

Zornitsa Bogomilova Kamburova1, Savelina Lubenova Popovska2, Katya Stefanova Kovacheva1, Krasimir Todorov Petrov2, Slavena Enkova Nikolova1.   

Abstract

Lynch syndrome (LS) is an autosomal dominant cancer syndrome. It can be caused by mutations of several genes, including MLH1, MSH2, MSH6, PMS2, MLH3 and MSH3, which are responsible for DNA mismatch repair, and LS affects 3-5% of patients with colorectal cancer (CRC). LS is associated with a high risk of cancer in several different locations, although the most commonly affected regions are the colon (20-70% risk), endometrium (15-70% risk), stomach (6-13% risk) and ovaries (4-12% risk). In the present report, the familial case of LS with a detected pathogenic variant in the MSH2 gene is described. The proband was a male who was diagnosed with CRC at the age of 25 years. Genealogy analysis revealed a total of seven affected relatives (including the proband), one of whom (I degree relative, mother) had synchronous cancers (endometrial and ovarian) and five others (of II and III degree relation) had ovarian cancer. Genetic analysis using next generation sequencing detected a heterozygous germline mutation in the MSH2 gene (c.1386 + 1G >A) in the proband and his mother, confirming the diagnosis of LS. The results of the recommended genetic test in an asymptomatic relative of the proband (II degree relative, uncle), found the same familial mutation. Subsequent prophylactic colonoscopy of this relative revealed early stage CRC. The presented case confirms the need for specific genetic analysis, alongside genetic counseling, in hereditary cancer syndromes. Active genetic prophylaxis in patients with LS allows early detection of primary cancers in other locations, and pre-symptomatic genetic analysis of relatives is an option for early diagnosis.
Copyright © 2020, Spandidos Publications.

Entities:  

Keywords:  Lynch syndrome; colorectal cancer; endometrial cancer; genetic counseling; mismatch repair deficiency; ovarian cancer; pathogenic variant; splice site

Year:  2022        PMID: 35386111      PMCID: PMC8972306          DOI: 10.3892/br.2022.1522

Source DB:  PubMed          Journal:  Biomed Rep        ISSN: 2049-9434


  30 in total

1.  A National Cancer Institute Workshop on Hereditary Nonpolyposis Colorectal Cancer Syndrome: meeting highlights and Bethesda guidelines.

Authors:  M A Rodriguez-Bigas; C R Boland; S R Hamilton; D E Henson; J R Jass; P M Khan; H Lynch; M Perucho; T Smyrk; L Sobin; S Srivastava
Journal:  J Natl Cancer Inst       Date:  1997-12-03       Impact factor: 13.506

2.  Differences in histological features and PD-L1 expression between sporadic microsatellite instability and Lynch-syndrome-associated disease in Japanese patients with colorectal cancer.

Authors:  Rin Yamada; Tatsuro Yamaguchi; Takeru Iijima; Rika Wakaume; Misato Takao; Koichi Koizumi; Tsunekazu Hishima; Shin-Ichiro Horiguchi
Journal:  Int J Clin Oncol       Date:  2018-01-11       Impact factor: 3.402

3.  Comparative in silico analyses and experimental validation of novel splice site and missense mutations in the genes MLH1 and MSH2.

Authors:  Beate Betz; Stephan Theiss; Murat Aktas; Carolin Konermann; Timm O Goecke; Gabriela Möslein; Heiner Schaal; Brigitte Royer-Pokora
Journal:  J Cancer Res Clin Oncol       Date:  2010-01       Impact factor: 4.553

4.  Spectrum and frequencies of mutations in MSH2 and MLH1 identified in 1,721 German families suspected of hereditary nonpolyposis colorectal cancer.

Authors:  Elisabeth Mangold; Constanze Pagenstecher; Waltraut Friedl; Micaela Mathiak; Reinhard Buettner; Christoph Engel; Markus Loeffler; Elke Holinski-Feder; Yvonne Müller-Koch; Gisela Keller; Hans K Schackert; Stefan Krüger; Timm Goecke; Gabriela Moeslein; Matthias Kloor; Johannes Gebert; Erdmute Kunstmann; Karsten Schulmann; Josef Rüschoff; Peter Propping
Journal:  Int J Cancer       Date:  2005-09-20       Impact factor: 7.396

5.  Colorectal Cancer Stratification in the Routine Clinical Pathway: A District General Hospital Experience.

Authors:  Sarah Wedden; Keith Miller; Ian M Frayling; Teresa Thomas; Alina Chefani; Karolina Miller; Angela Hamblin; Jenny C Taylor; Corrado D'Arrigo
Journal:  Appl Immunohistochem Mol Morphol       Date:  2019-07

6.  Surveillance in hereditary nonpolyposis colorectal cancer: an international cooperative study of 165 families. The International Collaborative Group on HNPCC.

Authors:  H F Vasen; J P Mecklin; P Watson; J Utsunomiya; L Bertario; P Lynch; L B Svendsen; G Cristofaro; H Müller; P M Khan
Journal:  Dis Colon Rectum       Date:  1993-01       Impact factor: 4.585

7.  Recent discoveries in the molecular genetics of Lynch syndrome.

Authors:  C Richard Boland
Journal:  Fam Cancer       Date:  2016-07       Impact factor: 2.375

Review 8.  Novel Implications in Molecular Diagnosis of Lynch Syndrome.

Authors:  Raffaella Liccardo; Marina De Rosa; Paola Izzo; Francesca Duraturo
Journal:  Gastroenterol Res Pract       Date:  2017-01-29       Impact factor: 2.260

Review 9.  Diagnosis of Lynch Syndrome and Strategies to Distinguish Lynch-Related Tumors from Sporadic MSI/dMMR Tumors.

Authors:  Julie Leclerc; Catherine Vermaut; Marie-Pierre Buisine
Journal:  Cancers (Basel)       Date:  2021-01-26       Impact factor: 6.639

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