Category Ranking

98%

Total Visits

921

Avg Visit Duration

2 minutes

Citations

20

Article Abstract

Coronary artery disease (CAD) including myocardial infarction (MI) is the leading cause of death worldwide and is commonly caused by the interaction between genetic factors and environmental risks. Despite intensive efforts using linkage and candidate gene approaches, the genetic etiology for the majority of families with a multigenerational early CAD /MI predisposition is unknown.In this study, we used whole-exome sequencing of 10 individuals from 1 early MI family, in which 4 siblings were diagnosed with MI before the age of 55, to identify potential predisposing genes.We identified a mutation in the RECQL5 gene, 1 of the 5 members of the RECQ family which are involved in the maintenance of genomic stability. This novel mutation, which is a TG insert at position 73,626,918 on the 13 chromosome and occurs before the last nucleotide of the introns 11 acceptor splice site affecting splicing of RECQL5. RT-PCR suggested the control subject had a full-length mRNA including exon 12, but the patients with RECQL5 mutation had a shorter mRNA form involving splicing of exons 11 to 13 directly, with skipping of exon 12. Quantitative RT-PCR analysis of RECQL5 exon 12 demonstrated that individuals whose genotype is mutant homozygote had only trace amounts of mRNA containing this exon and the family members who carry the heterozygous genotype had a level at 48% to 55% of the control's level.These findings provide insight into both the pathogenesis of MI and the role of RECQL5 gene in human disease.

Download full-text PDF

Source
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC4748938PMC
http://dx.doi.org/10.1097/MD.0000000000002737DOI Listing

Publication Analysis

Top Keywords

recql5 mutation
8
myocardial infarction
8
recql5 gene
8
recql5
6
exome sequencing
4
family
4
sequencing family
4
family identifies
4
identifies recql5
4
mutation
4

Similar Publications

Recql5-Deficient Mice as a Model for Studying Chromoanagenesis Phenomena.

Methods Mol Biol

August 2025

Center for Education in Laboratory Animal Research, Chubu University, Kasugai, Aichi, Japan.

Complex chromosomal rearrangements (CCRs) present significant challenges and opportunities in cancer and congenital disease research. Reproducing these rearrangements experimentally in animal models has been challenging, limiting our insights into their mechanisms and impacts. Recql5 is a critical DNA helicase that participates in replication, transcription, and repair processes.

View Article and Find Full Text PDF

Nonhomologous end joining (NHEJ) is required for repairing DNA double strand breaks (DSBs) generated by the RAG endonuclease during lymphocyte antigen receptor gene assembly by V(D)J recombination. The ataxia telangiectasia-mutated (ATM) and DNA-dependent protein kinase catalytic subunit (DNA-PKcs) kinases regulate functionally redundant pathways required for NHEJ. Here, we report that loss of the senataxin helicase leads to a strong defect in RAG DSB repair upon inactivation of DNA-PKcs.

View Article and Find Full Text PDF

The development of multiple primary tumors is one of the hallmarks of hereditary cancer. The phenotypic presentation of individuals with multiple primary tumors is often heterogeneous, which hampers the establishment of a genetic diagnosis. The absence of a genetic diagnosis may lead to inappropriate surveillance advices and treatment choices.

View Article and Find Full Text PDF

A double primary colorectal cancer (CRC) in a familial setting signals a high risk of CRC. In order to identify novel CRC susceptibility genes, we whole-exome sequenced germline DNA from nine persons with a double primary CRC and a family history of CRC. The detected variants were processed by bioinformatics filtering and prioritization, including STRING protein-protein interaction and pathway analysis.

View Article and Find Full Text PDF
Article Synopsis
  • Non-homologous end joining (NHEJ) is a crucial process for fixing DNA double strand breaks during the V(D)J recombination in immune cells.
  • The study finds that the loss of the senataxin helicase leads to difficulties in repairing these breaks, particularly when the DNA-PKcs is inactive.
  • It also reveals that senataxin works closely with other helicases like RECQL5 and translocases like HLTF, suggesting multiple backup systems are in place for effective NHEJ repair involving ATM and DNA-PKcs.
View Article and Find Full Text PDF