Cargando…
Clinical Considerations for a Family with Dilated Cardiomyopathy, Sudden Cardiac Death, and a Novel TTN Frameshift Mutation
Dilated cardiomyopathy (DCM) is the leading indication for heart transplantation. TTN gene truncating mutations account for about 25% of familial DCM cases and for 18% of sporadic DCM cases. The clinical relevance of specific variants in TTN has been difficult to determine because of the sheer size...
Autores principales: | , , , , , , , , , , |
---|---|
Formato: | Online Artículo Texto |
Lenguaje: | English |
Publicado: |
MDPI
2021
|
Materias: | |
Acceso en línea: | https://www.ncbi.nlm.nih.gov/pmc/articles/PMC7826882/ https://www.ncbi.nlm.nih.gov/pubmed/33445410 http://dx.doi.org/10.3390/ijms22020670 |
_version_ | 1783640626596675584 |
---|---|
author | Micaglio, Emanuele Monasky, Michelle M. Bernardini, Andrea Mecarocci, Valerio Borrelli, Valeria Ciconte, Giuseppe Locati, Emanuela T. Piccoli, Marco Ghiroldi, Andrea Anastasia, Luigi Pappone, Carlo |
author_facet | Micaglio, Emanuele Monasky, Michelle M. Bernardini, Andrea Mecarocci, Valerio Borrelli, Valeria Ciconte, Giuseppe Locati, Emanuela T. Piccoli, Marco Ghiroldi, Andrea Anastasia, Luigi Pappone, Carlo |
author_sort | Micaglio, Emanuele |
collection | PubMed |
description | Dilated cardiomyopathy (DCM) is the leading indication for heart transplantation. TTN gene truncating mutations account for about 25% of familial DCM cases and for 18% of sporadic DCM cases. The clinical relevance of specific variants in TTN has been difficult to determine because of the sheer size of the protein for which TTN encodes, as well as existing extensive genetic variation. Clinicians should communicate novel clinically-relevant variants and genotype–phenotype associations, so that animal studies evaluating the molecular mechanisms are always conducted with a focus on clinical significance. In the present study, we report for the first time the novel truncating heterozygous variant NM_001256850.1:c.72777_72783del (p.Phe24259Leufs*51) in the TTN gene and its association with DCM in a family with sudden death. This variant occurs in the A-band region of the sarcomere, in a known mutational hotspot of the gene. Truncating titin variants that occur in this region are the most common cause of DCM and have been rarely reported in asymptomatic individuals, differently from other pathogenic TTN gene variants. Further studies are warranted to better understand this particular clinically-relevant variant. |
format | Online Article Text |
id | pubmed-7826882 |
institution | National Center for Biotechnology Information |
language | English |
publishDate | 2021 |
publisher | MDPI |
record_format | MEDLINE/PubMed |
spelling | pubmed-78268822021-01-25 Clinical Considerations for a Family with Dilated Cardiomyopathy, Sudden Cardiac Death, and a Novel TTN Frameshift Mutation Micaglio, Emanuele Monasky, Michelle M. Bernardini, Andrea Mecarocci, Valerio Borrelli, Valeria Ciconte, Giuseppe Locati, Emanuela T. Piccoli, Marco Ghiroldi, Andrea Anastasia, Luigi Pappone, Carlo Int J Mol Sci Case Report Dilated cardiomyopathy (DCM) is the leading indication for heart transplantation. TTN gene truncating mutations account for about 25% of familial DCM cases and for 18% of sporadic DCM cases. The clinical relevance of specific variants in TTN has been difficult to determine because of the sheer size of the protein for which TTN encodes, as well as existing extensive genetic variation. Clinicians should communicate novel clinically-relevant variants and genotype–phenotype associations, so that animal studies evaluating the molecular mechanisms are always conducted with a focus on clinical significance. In the present study, we report for the first time the novel truncating heterozygous variant NM_001256850.1:c.72777_72783del (p.Phe24259Leufs*51) in the TTN gene and its association with DCM in a family with sudden death. This variant occurs in the A-band region of the sarcomere, in a known mutational hotspot of the gene. Truncating titin variants that occur in this region are the most common cause of DCM and have been rarely reported in asymptomatic individuals, differently from other pathogenic TTN gene variants. Further studies are warranted to better understand this particular clinically-relevant variant. MDPI 2021-01-12 /pmc/articles/PMC7826882/ /pubmed/33445410 http://dx.doi.org/10.3390/ijms22020670 Text en © 2021 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 | Case Report Micaglio, Emanuele Monasky, Michelle M. Bernardini, Andrea Mecarocci, Valerio Borrelli, Valeria Ciconte, Giuseppe Locati, Emanuela T. Piccoli, Marco Ghiroldi, Andrea Anastasia, Luigi Pappone, Carlo Clinical Considerations for a Family with Dilated Cardiomyopathy, Sudden Cardiac Death, and a Novel TTN Frameshift Mutation |
title | Clinical Considerations for a Family with Dilated Cardiomyopathy, Sudden Cardiac Death, and a Novel TTN Frameshift Mutation |
title_full | Clinical Considerations for a Family with Dilated Cardiomyopathy, Sudden Cardiac Death, and a Novel TTN Frameshift Mutation |
title_fullStr | Clinical Considerations for a Family with Dilated Cardiomyopathy, Sudden Cardiac Death, and a Novel TTN Frameshift Mutation |
title_full_unstemmed | Clinical Considerations for a Family with Dilated Cardiomyopathy, Sudden Cardiac Death, and a Novel TTN Frameshift Mutation |
title_short | Clinical Considerations for a Family with Dilated Cardiomyopathy, Sudden Cardiac Death, and a Novel TTN Frameshift Mutation |
title_sort | clinical considerations for a family with dilated cardiomyopathy, sudden cardiac death, and a novel ttn frameshift mutation |
topic | Case Report |
url | https://www.ncbi.nlm.nih.gov/pmc/articles/PMC7826882/ https://www.ncbi.nlm.nih.gov/pubmed/33445410 http://dx.doi.org/10.3390/ijms22020670 |
work_keys_str_mv | AT micaglioemanuele clinicalconsiderationsforafamilywithdilatedcardiomyopathysuddencardiacdeathandanovelttnframeshiftmutation AT monaskymichellem clinicalconsiderationsforafamilywithdilatedcardiomyopathysuddencardiacdeathandanovelttnframeshiftmutation AT bernardiniandrea clinicalconsiderationsforafamilywithdilatedcardiomyopathysuddencardiacdeathandanovelttnframeshiftmutation AT mecaroccivalerio clinicalconsiderationsforafamilywithdilatedcardiomyopathysuddencardiacdeathandanovelttnframeshiftmutation AT borrellivaleria clinicalconsiderationsforafamilywithdilatedcardiomyopathysuddencardiacdeathandanovelttnframeshiftmutation AT cicontegiuseppe clinicalconsiderationsforafamilywithdilatedcardiomyopathysuddencardiacdeathandanovelttnframeshiftmutation AT locatiemanuelat clinicalconsiderationsforafamilywithdilatedcardiomyopathysuddencardiacdeathandanovelttnframeshiftmutation AT piccolimarco clinicalconsiderationsforafamilywithdilatedcardiomyopathysuddencardiacdeathandanovelttnframeshiftmutation AT ghiroldiandrea clinicalconsiderationsforafamilywithdilatedcardiomyopathysuddencardiacdeathandanovelttnframeshiftmutation AT anastasialuigi clinicalconsiderationsforafamilywithdilatedcardiomyopathysuddencardiacdeathandanovelttnframeshiftmutation AT papponecarlo clinicalconsiderationsforafamilywithdilatedcardiomyopathysuddencardiacdeathandanovelttnframeshiftmutation |