Cargando…

Clinical Significance of Variants in the TTN Gene in a Large Cohort of Patients With Sporadic Dilated Cardiomyopathy

Background: Mutations in the TTN gene are the most common causes of dilated cardiomyopathy (DCM). The clinical significance of TTN gene variants remains inadequately understood. Methods: Whole-exome sequencing and phenotypic characterisation were performed, and patients were followed up for a median...

Descripción completa

Detalles Bibliográficos
Autores principales: Xiao, Lei, Li, Chenze, Sun, Yang, Chen, Yanghui, Wei, Haoran, Hu, Dong, Yu, Ting, Li, Xianqing, Jin, Li, Shi, Leming, Marian, Ali J., Wang, Dao Wen
Formato: Online Artículo Texto
Lenguaje:English
Publicado: Frontiers Media S.A. 2021
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC8120103/
https://www.ncbi.nlm.nih.gov/pubmed/33996946
http://dx.doi.org/10.3389/fcvm.2021.657689
_version_ 1783691984317186048
author Xiao, Lei
Li, Chenze
Sun, Yang
Chen, Yanghui
Wei, Haoran
Hu, Dong
Yu, Ting
Li, Xianqing
Jin, Li
Shi, Leming
Marian, Ali J.
Wang, Dao Wen
author_facet Xiao, Lei
Li, Chenze
Sun, Yang
Chen, Yanghui
Wei, Haoran
Hu, Dong
Yu, Ting
Li, Xianqing
Jin, Li
Shi, Leming
Marian, Ali J.
Wang, Dao Wen
author_sort Xiao, Lei
collection PubMed
description Background: Mutations in the TTN gene are the most common causes of dilated cardiomyopathy (DCM). The clinical significance of TTN gene variants remains inadequately understood. Methods: Whole-exome sequencing and phenotypic characterisation were performed, and patients were followed up for a median of 44 months. Results: We analyzed the association of the TTN variants with the clinical outcomes in a prospective study of 1,041 patients with sporadic DCM. TTN truncating variants (tTTN) were detected in 120 (11.5%) patients as compared with 2.4/10,000 East Asian populations in the Genome Aggregation Database (GnomAD; p < 0.0001). Pathogenic TTN missense variants were also enriched in DCM as compared with the GnomAD populations (27.6 vs. 5.9%, p < 0.0001). DCM patients with tTTN had a lower left ventricular ejection fraction (28.89 ± 8.72 vs. 31.81 ± 9.97, p = 0.002) and a lower frequency of the left bundle branch block (3.3 vs. 11.3%, p = 0.011) than those without or with mutations in other known causal genes (OCG). However, tTTN were not associated with the composite primary endpoint of cardiac death and heart transplantation during the follow-up period [adjusted hazard ratio (HR): 0.912; 95% confidence interval: 0.464–1.793; p = 0.790]. There was also no sex-dependent effect. Concomitant tTTN and pathogenic variants in OCG were present in only eight DCM patients and did not affect the outcome. Conclusion: The phenotype of DCM caused by tTTN, major causes of sporadic DCM, is not distinctly different from those caused by other causal genes for DCM.
format Online
Article
Text
id pubmed-8120103
institution National Center for Biotechnology Information
language English
publishDate 2021
publisher Frontiers Media S.A.
record_format MEDLINE/PubMed
spelling pubmed-81201032021-05-15 Clinical Significance of Variants in the TTN Gene in a Large Cohort of Patients With Sporadic Dilated Cardiomyopathy Xiao, Lei Li, Chenze Sun, Yang Chen, Yanghui Wei, Haoran Hu, Dong Yu, Ting Li, Xianqing Jin, Li Shi, Leming Marian, Ali J. Wang, Dao Wen Front Cardiovasc Med Cardiovascular Medicine Background: Mutations in the TTN gene are the most common causes of dilated cardiomyopathy (DCM). The clinical significance of TTN gene variants remains inadequately understood. Methods: Whole-exome sequencing and phenotypic characterisation were performed, and patients were followed up for a median of 44 months. Results: We analyzed the association of the TTN variants with the clinical outcomes in a prospective study of 1,041 patients with sporadic DCM. TTN truncating variants (tTTN) were detected in 120 (11.5%) patients as compared with 2.4/10,000 East Asian populations in the Genome Aggregation Database (GnomAD; p < 0.0001). Pathogenic TTN missense variants were also enriched in DCM as compared with the GnomAD populations (27.6 vs. 5.9%, p < 0.0001). DCM patients with tTTN had a lower left ventricular ejection fraction (28.89 ± 8.72 vs. 31.81 ± 9.97, p = 0.002) and a lower frequency of the left bundle branch block (3.3 vs. 11.3%, p = 0.011) than those without or with mutations in other known causal genes (OCG). However, tTTN were not associated with the composite primary endpoint of cardiac death and heart transplantation during the follow-up period [adjusted hazard ratio (HR): 0.912; 95% confidence interval: 0.464–1.793; p = 0.790]. There was also no sex-dependent effect. Concomitant tTTN and pathogenic variants in OCG were present in only eight DCM patients and did not affect the outcome. Conclusion: The phenotype of DCM caused by tTTN, major causes of sporadic DCM, is not distinctly different from those caused by other causal genes for DCM. Frontiers Media S.A. 2021-04-30 /pmc/articles/PMC8120103/ /pubmed/33996946 http://dx.doi.org/10.3389/fcvm.2021.657689 Text en Copyright © 2021 Xiao, Li, Sun, Chen, Wei, Hu, Yu, Li, Jin, Shi, Marian and Wang. https://creativecommons.org/licenses/by/4.0/This is an open-access article distributed under the terms of the Creative Commons Attribution License (CC BY). The use, distribution or reproduction in other forums is permitted, provided the original author(s) and the copyright owner(s) are credited and that the original publication in this journal is cited, in accordance with accepted academic practice. No use, distribution or reproduction is permitted which does not comply with these terms.
spellingShingle Cardiovascular Medicine
Xiao, Lei
Li, Chenze
Sun, Yang
Chen, Yanghui
Wei, Haoran
Hu, Dong
Yu, Ting
Li, Xianqing
Jin, Li
Shi, Leming
Marian, Ali J.
Wang, Dao Wen
Clinical Significance of Variants in the TTN Gene in a Large Cohort of Patients With Sporadic Dilated Cardiomyopathy
title Clinical Significance of Variants in the TTN Gene in a Large Cohort of Patients With Sporadic Dilated Cardiomyopathy
title_full Clinical Significance of Variants in the TTN Gene in a Large Cohort of Patients With Sporadic Dilated Cardiomyopathy
title_fullStr Clinical Significance of Variants in the TTN Gene in a Large Cohort of Patients With Sporadic Dilated Cardiomyopathy
title_full_unstemmed Clinical Significance of Variants in the TTN Gene in a Large Cohort of Patients With Sporadic Dilated Cardiomyopathy
title_short Clinical Significance of Variants in the TTN Gene in a Large Cohort of Patients With Sporadic Dilated Cardiomyopathy
title_sort clinical significance of variants in the ttn gene in a large cohort of patients with sporadic dilated cardiomyopathy
topic Cardiovascular Medicine
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC8120103/
https://www.ncbi.nlm.nih.gov/pubmed/33996946
http://dx.doi.org/10.3389/fcvm.2021.657689
work_keys_str_mv AT xiaolei clinicalsignificanceofvariantsinthettngeneinalargecohortofpatientswithsporadicdilatedcardiomyopathy
AT lichenze clinicalsignificanceofvariantsinthettngeneinalargecohortofpatientswithsporadicdilatedcardiomyopathy
AT sunyang clinicalsignificanceofvariantsinthettngeneinalargecohortofpatientswithsporadicdilatedcardiomyopathy
AT chenyanghui clinicalsignificanceofvariantsinthettngeneinalargecohortofpatientswithsporadicdilatedcardiomyopathy
AT weihaoran clinicalsignificanceofvariantsinthettngeneinalargecohortofpatientswithsporadicdilatedcardiomyopathy
AT hudong clinicalsignificanceofvariantsinthettngeneinalargecohortofpatientswithsporadicdilatedcardiomyopathy
AT yuting clinicalsignificanceofvariantsinthettngeneinalargecohortofpatientswithsporadicdilatedcardiomyopathy
AT lixianqing clinicalsignificanceofvariantsinthettngeneinalargecohortofpatientswithsporadicdilatedcardiomyopathy
AT jinli clinicalsignificanceofvariantsinthettngeneinalargecohortofpatientswithsporadicdilatedcardiomyopathy
AT shileming clinicalsignificanceofvariantsinthettngeneinalargecohortofpatientswithsporadicdilatedcardiomyopathy
AT marianalij clinicalsignificanceofvariantsinthettngeneinalargecohortofpatientswithsporadicdilatedcardiomyopathy
AT wangdaowen clinicalsignificanceofvariantsinthettngeneinalargecohortofpatientswithsporadicdilatedcardiomyopathy