Literature DB >> 30935706

A Novel Truncating Variant in FLNC-Encoded Filamin C May Serve as a Proarrhythmic Genetic Substrate for Arrhythmogenic Bileaflet Mitral Valve Prolapse Syndrome.

Sahej Bains1, David J Tester2, Samuel J Asirvatham3, Peter A Noseworthy3, Michael J Ackerman4, John R Giudicessi5.   

Abstract

A 51-year-old man with a long-standing history of bileaflet mitral valve prolapse accompanied by mitral annular disjunction and mild mitral regurgitation presented for evaluation of increasingly frequent palpitations. Ambulatory Holter monitoring, cardiac magnetic resonance imaging, serial transthoracic echocardiography, and diagnostic electrophysiology studies were consistent with a diagnosis of arrhythmogenic bileaflet mitral valve prolapse syndrome. Because of the presence of a similar phenotype in the proband's mother, brother, and maternal aunt, research-based whole exome sequencing was pursued and a novel truncating variant (p.Trp34*-FLNC) in the cardiomyopathy-causative FLNC-encoded filamin C unearthed that cosegregated with disease. Unexpectedly, these observations provide the first evidence that a heritable proarrhythmic genetic substrate (ie, FLNC haploinsufficiency-mediated weakening of cell-cell adhesion) may underlie, at least in part, some cases of arrhythmogenic mitral valve prolapse syndrome.
Copyright © 2018 Mayo Foundation for Medical Education and Research. Published by Elsevier Inc. All rights reserved.

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Year:  2019        PMID: 30935706     DOI: 10.1016/j.mayocp.2018.11.028

Source DB:  PubMed          Journal:  Mayo Clin Proc        ISSN: 0025-6196            Impact factor:   7.616


  12 in total

1.  Long-term outcomes of ablation for ventricular arrhythmias in mitral valve prolapse.

Authors:  Paul J Marano; Lisa J Lim; Jose M Sanchez; Raza Alvi; Gregory Nah; Nitish Badhwar; Edward P Gerstenfeld; Zian H Tseng; Gregory M Marcus; Francesca N Delling
Journal:  J Interv Card Electrophysiol       Date:  2020-06-06       Impact factor: 1.900

2.  Malignant Arrhythmic Mitral Valve Prolapse: A Continuum of Clinical Challenges from Diagnosis to Risk Stratification and Patient Management.

Authors:  Idit Yedidya; Aniek L van Wijngaarden; Nina Ajmone Marsan
Journal:  J Cardiovasc Dev Dis       Date:  2020-12-29

Review 3.  Cardiac Filaminopathies: Illuminating the Divergent Role of Filamin C Mutations in Human Cardiomyopathy.

Authors:  Matthias Eden; Norbert Frey
Journal:  J Clin Med       Date:  2021-02-04       Impact factor: 4.241

Review 4.  The Outcome of Ventricular Arrhythmias Associated With Mitral Valve Prolapse After Catheter Ablation: A Systematic Review and Meta-Analysis.

Authors:  Kevin Wibawa; Ignatius Ivan; Giovanni Jessica; Denio Ridjab
Journal:  Cureus       Date:  2021-12-09

5.  Prevalence and potential genetic determinants of young sudden unexplained death victims with suspected arrhythmogenic mitral valve prolapse syndrome.

Authors:  John R Giudicessi; Joseph J Maleszewski; David J Tester; Michael J Ackerman
Journal:  Heart Rhythm O2       Date:  2021-07-22

Review 6.  Developing a Mechanistic Approach to Sudden Death Prevention in Mitral Valve Prolapse.

Authors:  Brian P Kelley; Abdul Mateen Chaudry; Faisal F Syed
Journal:  J Clin Med       Date:  2022-02-26       Impact factor: 4.241

7.  Clinical exome sequencing revealed that FLNC variants contribute to the early diagnosis of cardiomyopathies in infant patients.

Authors:  Feifan Xiao; Qiufen Wei; Bingbing Wu; Xu Liu; Aiyao Mading; Lin Yang; Yan Li; Fang Liu; Xinnian Pan; Huijun Wang
Journal:  Transl Pediatr       Date:  2020-02

8.  A mutation update for the FLNC gene in myopathies and cardiomyopathies.

Authors:  Job A J Verdonschot; Els K Vanhoutte; Godelieve R F Claes; Apollonia T J M Helderman-van den Enden; Janneke G J Hoeijmakers; Debby M E I Hellebrekers; Amber de Haan; Imke Christiaans; Ronald H Lekanne Deprez; Hanne M Boen; Emeline M van Craenenbroeck; Bart L Loeys; Yvonne M Hoedemaekers; Carlo Marcelis; Marlies Kempers; Esther Brusse; Jaap I van Waning; Annette F Baas; Dennis Dooijes; Folkert W Asselbergs; Daniela Q C M Barge-Schaapveld; Pieter Koopman; Arthur van den Wijngaard; Stephane R B Heymans; Ingrid P C Krapels; Han G Brunner
Journal:  Hum Mutat       Date:  2020-03-20       Impact factor: 4.878

Review 9.  Structure and Function of Filamin C in the Muscle Z-Disc.

Authors:  Zhenfeng Mao; Fumihiko Nakamura
Journal:  Int J Mol Sci       Date:  2020-04-13       Impact factor: 5.923

Review 10.  The Role of Z-disc Proteins in Myopathy and Cardiomyopathy.

Authors:  Kirsty Wadmore; Amar J Azad; Katja Gehmlich
Journal:  Int J Mol Sci       Date:  2021-03-17       Impact factor: 6.208

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