Literature DB >> 31816043

Progressive cardiac arrhythmias and ECG abnormalities in the Huntington's disease BACHD mouse model.

Yujie Zhu1, Isaac Shamblin1, Efrain Rodriguez2, Grace E Salzer1, Lita Araysi1, Katherine A Margolies1, Ganesh V Halade1, Silvio H Litovsky3, Steven Pogwizd1, Michelle Gray2, Sabine Huke1.   

Abstract

Huntington's disease (HD) is a dominantly inherited neurodegenerative disease. There is accumulating evidence that HD patients have increased prevalence of conduction abnormalities and compromised sinoatrial node function which could lead to increased risk for arrhythmia. We used mutant Huntingtin (mHTT) expressing bacterial artificial chromosome Huntington's disease mice to determine if they exhibit electrocardiogram (ECG) abnormalities involving cardiac conduction that are known to increase risk of sudden arrhythmic death in humans. We obtained surface ECGs and analyzed arrhythmia susceptibility; we observed prolonged QRS duration, increases in PVCs as well as PACs. Abnormal histological and structural changes that could lead to cardiac conduction system dysfunction were seen. Finally, we observed decreases in desmosomal proteins, plakophilin-2 and desmoglein-2, which have been reported to cause cardiac arrhythmias and reduced conduction. Our study indicates that mHTT could cause progressive cardiac conduction system pathology that could increase the susceptibility to arrhythmias and sudden cardiac death in HD patients.
© The Author(s) 2019. Published by Oxford University Press. All rights reserved. For Permissions, please email: journals.permissions@oup.com.

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Year:  2020        PMID: 31816043      PMCID: PMC7015842          DOI: 10.1093/hmg/ddz295

Source DB:  PubMed          Journal:  Hum Mol Genet        ISSN: 0964-6906            Impact factor:   6.150


  77 in total

1.  Intercalated discs and arrhythmogenic cardiomyopathy.

Authors:  Alessandra Rampazzo; Martina Calore; Jolanda van Hengel; Frans van Roy
Journal:  Circ Cardiovasc Genet       Date:  2014-12

2.  Huntingtin Aggregates and Mitochondrial Pathology in Skeletal Muscle but not Heart of Late-Stage R6/2 Mice.

Authors:  Kerstin Kojer; Tanja Hering; Chantal Bazenet; Andreas Weiss; Frank Herrmann; Jan-Willem Taanman; Michael Orth
Journal:  J Huntingtons Dis       Date:  2019

3.  Conditions associated with Huntington's disease at death. A case-control study.

Authors:  D J Lanska; M J Lanska; L Lavine; B S Schoenberg
Journal:  Arch Neurol       Date:  1988-08

4.  Biological and clinical changes in premanifest and early stage Huntington's disease in the TRACK-HD study: the 12-month longitudinal analysis.

Authors:  Sarah J Tabrizi; Rachael I Scahill; Alexandra Durr; Raymund Ac Roos; Blair R Leavitt; Rebecca Jones; G Bernhard Landwehrmeyer; Nick C Fox; Hans Johnson; Stephen L Hicks; Christopher Kennard; David Craufurd; Chris Frost; Douglas R Langbehn; Ralf Reilmann; Julie C Stout
Journal:  Lancet Neurol       Date:  2010-12-02       Impact factor: 44.182

5.  Huntingtin localization in brains of normal and Huntington's disease patients.

Authors:  E Sapp; C Schwarz; K Chase; P G Bhide; A B Young; J Penney; J P Vonsattel; N Aronin; M DiFiglia
Journal:  Ann Neurol       Date:  1997-10       Impact factor: 10.422

6.  Intercalated disc abnormalities, reduced Na(+) current density, and conduction slowing in desmoglein-2 mutant mice prior to cardiomyopathic changes.

Authors:  Stefania Rizzo; Elisabeth M Lodder; Arie O Verkerk; Rianne Wolswinkel; Leander Beekman; Kalliopi Pilichou; Cristina Basso; Carol Ann Remme; Gaetano Thiene; Connie R Bezzina
Journal:  Cardiovasc Res       Date:  2012-07-03       Impact factor: 10.787

7.  Autonomic symptoms in patients and pre-manifest mutation carriers of Huntington's disease.

Authors:  N A Aziz; G V Anguelova; J Marinus; J G van Dijk; R A C Roos
Journal:  Eur J Neurol       Date:  2010-02-24       Impact factor: 6.089

8.  Characterization of cardiac conduction system abnormalities in mice with targeted disruption of Six5 gene.

Authors:  Hiroko Wakimoto; Colin T Maguire; Megan C Sherwood; Marcel M Vargas; Partha S Sarkar; Jennifer Han; Sita Reddy; Charles I Berul
Journal:  J Interv Card Electrophysiol       Date:  2002-10       Impact factor: 1.900

Review 9.  Skeletal muscle pathology in Huntington's disease.

Authors:  Daniel Zielonka; Izabela Piotrowska; Jerzy T Marcinkowski; Michal Mielcarek
Journal:  Front Physiol       Date:  2014-10-06       Impact factor: 4.566

10.  Overlap between age-at-onset and disease-progression determinants in Huntington disease.

Authors:  N Ahmad Aziz; Jorien M M van der Burg; Sarah J Tabrizi; G Bernhard Landwehrmeyer
Journal:  Neurology       Date:  2018-05-09       Impact factor: 9.910

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  2 in total

1.  Development and optimization of an in vivo electrocardiogram recording method and analysis program for adult zebrafish.

Authors:  ThuyVy Duong; Rebecca Rose; Adriana Blazeski; Noah Fine; Courtney E Woods; Joseph F Thole; Nona Sotoodehnia; Elsayed Z Soliman; Leslie Tung; Andrew S McCallion; Dan E Arking
Journal:  Dis Model Mech       Date:  2021-08-11       Impact factor: 5.758

2.  Targeted Genetic Reduction of Mutant Huntingtin Lessens Cardiac Pathology in the BACHD Mouse Model of Huntington's Disease.

Authors:  Saemi Park; Shu Hon Christopher Luk; Raj S Bains; Daniel S Whittaker; Emily Chiem; Maria C Jordan; Kenneth P Roos; Cristina A Ghiani; Christopher S Colwell
Journal:  Front Cardiovasc Med       Date:  2021-12-24
  2 in total

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