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...

Descripción completa

Detalles Bibliográficos
Autores principales: Micaglio, Emanuele, Monasky, Michelle M., Bernardini, Andrea, Mecarocci, Valerio, Borrelli, Valeria, Ciconte, Giuseppe, Locati, Emanuela T., Piccoli, Marco, Ghiroldi, Andrea, Anastasia, Luigi, Pappone, Carlo
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