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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...

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Autores principales: 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
Formato: Online Artículo Texto
Lenguaje:English
Publicado: MDPI 2020
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.
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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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