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Analysis of De Novo Mutations in Sporadic Cardiomyopathies Emphasizes Their Clinical Relevance and Points to Novel Candidate Genes
The vast majority of cardiomyopathies have an autosomal dominant inheritance; hence, genetic testing is typically offered to patients with a positive family history. A de novo mutation is a new germline mutation not inherited from either parent. The purpose of our study was to search for de novo mut...
Autores principales: | , , , , , , , , , , , , , , , , |
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Formato: | Online Artículo Texto |
Lenguaje: | English |
Publicado: |
MDPI
2020
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Materias: | |
Acceso en línea: | https://www.ncbi.nlm.nih.gov/pmc/articles/PMC7073782/ https://www.ncbi.nlm.nih.gov/pubmed/32013205 http://dx.doi.org/10.3390/jcm9020370 |
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author | Franaszczyk, Maria Truszkowska, Grazyna Chmielewski, Przemyslaw Rydzanicz, Malgorzata Kosinska, Joanna Rywik, Tomasz Biernacka, Anna Spiewak, Mateusz Kostrzewa, Grazyna Stepien-Wojno, Malgorzata Stawinski, Piotr Bilinska, Maria Krajewski, Pawel Zielinski, Tomasz Lutynska, Anna Bilinska, Zofia T. Ploski, Rafal |
author_facet | Franaszczyk, Maria Truszkowska, Grazyna Chmielewski, Przemyslaw Rydzanicz, Malgorzata Kosinska, Joanna Rywik, Tomasz Biernacka, Anna Spiewak, Mateusz Kostrzewa, Grazyna Stepien-Wojno, Malgorzata Stawinski, Piotr Bilinska, Maria Krajewski, Pawel Zielinski, Tomasz Lutynska, Anna Bilinska, Zofia T. Ploski, Rafal |
author_sort | Franaszczyk, Maria |
collection | PubMed |
description | The vast majority of cardiomyopathies have an autosomal dominant inheritance; hence, genetic testing is typically offered to patients with a positive family history. A de novo mutation is a new germline mutation not inherited from either parent. The purpose of our study was to search for de novo mutations in patients with cardiomyopathy and no evidence of the disease in the family. Using next-generation sequencing, we analyzed cardiomyopathy genes in 12 probands. In 8 (66.7%), we found de novo variants in known cardiomyopathy genes (TTN, DSP, SCN5A, TNNC1, TPM1, CRYAB, MYH7). In the remaining probands, the analysis was extended to whole exome sequencing in a trio (proband and parents). We found de novo variants in genes that, so far, were not associated with any disease (TRIB3, SLC2A6), a possible disease-causing biallelic genotype (APOBEC gene family), and a de novo mosaic variant without strong evidence of pathogenicity (UNC45A). The high prevalence of de novo mutations emphasizes that genetic screening is also indicated in cases of sporadic cardiomyopathy. Moreover, we have identified novel cardiomyopathy candidate genes that are likely to affect immunological function and/or reaction to stress that could be especially relevant in patients with disease onset associated with infection/infestation. |
format | Online Article Text |
id | pubmed-7073782 |
institution | National Center for Biotechnology Information |
language | English |
publishDate | 2020 |
publisher | MDPI |
record_format | MEDLINE/PubMed |
spelling | pubmed-70737822020-03-19 Analysis of De Novo Mutations in Sporadic Cardiomyopathies Emphasizes Their Clinical Relevance and Points to Novel Candidate Genes Franaszczyk, Maria Truszkowska, Grazyna Chmielewski, Przemyslaw Rydzanicz, Malgorzata Kosinska, Joanna Rywik, Tomasz Biernacka, Anna Spiewak, Mateusz Kostrzewa, Grazyna Stepien-Wojno, Malgorzata Stawinski, Piotr Bilinska, Maria Krajewski, Pawel Zielinski, Tomasz Lutynska, Anna Bilinska, Zofia T. Ploski, Rafal J Clin Med Article The vast majority of cardiomyopathies have an autosomal dominant inheritance; hence, genetic testing is typically offered to patients with a positive family history. A de novo mutation is a new germline mutation not inherited from either parent. The purpose of our study was to search for de novo mutations in patients with cardiomyopathy and no evidence of the disease in the family. Using next-generation sequencing, we analyzed cardiomyopathy genes in 12 probands. In 8 (66.7%), we found de novo variants in known cardiomyopathy genes (TTN, DSP, SCN5A, TNNC1, TPM1, CRYAB, MYH7). In the remaining probands, the analysis was extended to whole exome sequencing in a trio (proband and parents). We found de novo variants in genes that, so far, were not associated with any disease (TRIB3, SLC2A6), a possible disease-causing biallelic genotype (APOBEC gene family), and a de novo mosaic variant without strong evidence of pathogenicity (UNC45A). The high prevalence of de novo mutations emphasizes that genetic screening is also indicated in cases of sporadic cardiomyopathy. Moreover, we have identified novel cardiomyopathy candidate genes that are likely to affect immunological function and/or reaction to stress that could be especially relevant in patients with disease onset associated with infection/infestation. MDPI 2020-01-29 /pmc/articles/PMC7073782/ /pubmed/32013205 http://dx.doi.org/10.3390/jcm9020370 Text en © 2020 by the authors. Licensee MDPI, Basel, Switzerland. This article is an open access article distributed under the terms and conditions of the Creative Commons Attribution (CC BY) license (http://creativecommons.org/licenses/by/4.0/). |
spellingShingle | Article Franaszczyk, Maria Truszkowska, Grazyna Chmielewski, Przemyslaw Rydzanicz, Malgorzata Kosinska, Joanna Rywik, Tomasz Biernacka, Anna Spiewak, Mateusz Kostrzewa, Grazyna Stepien-Wojno, Malgorzata Stawinski, Piotr Bilinska, Maria Krajewski, Pawel Zielinski, Tomasz Lutynska, Anna Bilinska, Zofia T. Ploski, Rafal Analysis of De Novo Mutations in Sporadic Cardiomyopathies Emphasizes Their Clinical Relevance and Points to Novel Candidate Genes |
title | Analysis of De Novo Mutations in Sporadic Cardiomyopathies Emphasizes Their Clinical Relevance and Points to Novel Candidate Genes |
title_full | Analysis of De Novo Mutations in Sporadic Cardiomyopathies Emphasizes Their Clinical Relevance and Points to Novel Candidate Genes |
title_fullStr | Analysis of De Novo Mutations in Sporadic Cardiomyopathies Emphasizes Their Clinical Relevance and Points to Novel Candidate Genes |
title_full_unstemmed | Analysis of De Novo Mutations in Sporadic Cardiomyopathies Emphasizes Their Clinical Relevance and Points to Novel Candidate Genes |
title_short | Analysis of De Novo Mutations in Sporadic Cardiomyopathies Emphasizes Their Clinical Relevance and Points to Novel Candidate Genes |
title_sort | analysis of de novo mutations in sporadic cardiomyopathies emphasizes their clinical relevance and points to novel candidate genes |
topic | Article |
url | https://www.ncbi.nlm.nih.gov/pmc/articles/PMC7073782/ https://www.ncbi.nlm.nih.gov/pubmed/32013205 http://dx.doi.org/10.3390/jcm9020370 |
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