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Genome-wide copy neutral LOH is infrequent in familial and sporadic microsatellite unstable carcinomas

Authors :
Anneke Middeldorp
Tom van Wezel
Frederik J. Hes
Ronald van Eijk
Hans Morreau
Jan Oosting
Carli M. J. Tops
Heleen M. van der Klift
Marjo van Puijenbroek
Juul T. Wijnen
Hans F. A. Vasen
Clinical sciences
Medical Genetics
Source :
Familial Cancer. 7:319-330
Publication Year :
2008
Publisher :
Springer Science and Business Media LLC, 2008.

Abstract

Mismatch repair deficiency in tumors can result from germ line mutations in one of the mismatch repair (MMR) genes (MLH1, MSH2, MSH6 and PMS2), or from sporadic promoter hypermethylation of MLH1. The role of unclassified variants (UVs) in MMR genes is subject to debate. To establish the extend of chromosomal instability and copy neutral loss of heterozygosity (cnLOH), we analyzed 41 archival microsatellite unstable carcinomas, mainly colon cancer, from 23 patients with pathogenic MMR mutations, from eight patients with UVs in one of the MMR genes and 10 cases with MLH1 promoter hypermethylation. We assessed genome wide copy number abnormalities and cnLOH using SNP arrays. SNP arrays overcome the problems of detecting LOH due to instability of polymorphic microsatellite markers. All carcinomas showed relatively few chromosomal aberrations. Also cnLOH was infrequent and in Lynch syndrome carcinomas usually confined to the locus harbouring pathogenic mutations in MLH1, MSH2 or PMS2 In the carcinomas from the MMR-UV carriers such cnLOH was less common and in the carcinomas with MLH1 promoter hypermethylation no cnLOH at MLH1 occurred. MSI-H carcinomas of most MMR-UV carriers present on average with more aberrations compared to the carcinomas from pathogenic MMR mutation carriers, suggesting that another possible pathogenic MMR mutation had not been missed. The approach we describe here shows to be an excellent way to study genome-wide cnLOH in archival mismatch repair deficient tumors.

Details

ISSN :
15737292 and 13899600
Volume :
7
Database :
OpenAIRE
Journal :
Familial Cancer
Accession number :
edsair.doi.dedup.....ab33a1c23b4a8bf390c97746c0c93493
Full Text :
https://doi.org/10.1007/s10689-008-9194-8