Evidence map›Paper›PMID 34990403›Full record

ArticleJCI insight2022

The Purkinje-myocardial junction is the anatomic origin of ventricular arrhythmia in CPVT.

Daniel J Blackwell, Michela Faggioni, Matthew J Wleklinski, Nieves Gomez-Hurtado, Raghav Venkataraman, Chelsea E Gibbs, Franz J Baudenbacher, Shiaoching Gong, Glenn I Fishman, Patrick M Boyle and 2 more

Open access · goldAbstract read
In one paragraph

Article in JCI insight, 2022. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 26 papers.

0numbers the graph read from it
0cells of the map it votes in
26citing papers in PubMed
4.8field-weighted citation impact, top 4% of its field
1 · What the graph read from it

What it found

Each row is one number read from the abstract, on the scale the paper reported it, with its interval. Left of the dashed line favours the treatment, right favours the comparator. Under each row is the sentence it came from. New to these charts? A ten-minute tutorial.

The abstract states no effect estimate the extractor could read, or names no intervention and outcome on the map, so this paper lights no cell and moves no belief. It is still indexed, cited and linked below.

2 · The registry

The trial behind it

Trials whose registry record cites this paper, or whose number appears in the abstract. A trial that started after this paper was published is citing it as background, not reporting it.

Neither the registry nor the abstract names a trial number. If this is a trial report, that itself is worth knowing.

3 · Its place in the literature

Who cites it

26 citing papers in PubMed, 33 citations in OpenAlex.

  1. Calmodulin mutation N54I causes autosomal dominant CPVT in mice.bioRxiv : the preprint server for biology · 2026
    Article
  2. An RYR2-interacting fragment of MYBPC3 mitigates arrhythmia in human iPSC-CM and mouse models of CPVT.Molecular therapy : the journal of the American Society of Gene Therapy · 2026
    Article
  3. Review
  4. Ca2+ signalling in cardiac muscle: the importance of balances.Europace : European pacing, arrhythmias, and cardiac electrophysiology : journal of the working groups on cardiac pacing, arrhythmias, and cardiac cellular electrophysiology of the European Society of Cardiology · 2026
    Review
  5. Article
  6. Article
  7. Review
  8. Article
  9. Review
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  11. Article
  12. Stem cell models of inherited arrhythmias.Nature cardiovascular research · 2024
    Review
  13. Management of patients with an electrical storm or clustered ventricular arrhythmias: a clinical consensus statement of the European Heart Rhythm Association of the ESC-endorsed by the Asia-Pacific Heart Rhythm Society, Heart Rhythm Society, and Latin-American Heart Rhythm Society.Europace : European pacing, arrhythmias, and cardiac electrophysiology : journal of the working groups on cardiac pacing, arrhythmias, and cardiac cellular electrophysiology of the European Society of Cardiology · 2024
    Article
  14. Fine tuning contractility: atrial sarcomere function in health and disease.American journal of physiology. Heart and circulatory physiology · 2024
    Review
  15. Molecular pharmacology · 2024
    Article
  16. Article
  17. Article
  18. Article
  19. ELUCIDATING THE ROLE OF THE HIS-PURKINJE SYSTEM DURING LONG QT MEDIATED ARRHYTHMIAS.Annual Modeling and Simulation Conference (ANNSIM). Annual Modeling and Simulation Conference (Online) · 2023
    Article
  20. Article
4 · The record

Corrections and comments

PubMed lists nothing against this paper. Absence here is not a guarantee, only a check that was made.

5 · Who and what money

Authors and funding

12 authors at 5 institutions in 2 countries.

Daniel J BlackwellVanderbilt Center for Arrhythmia Research and Therapeutics, Division of Clinical Pharmacology, Department of Medicine, Vanderbilt University School of Medicine, Nashville, Tennessee, USA.
Michela FaggioniVanderbilt Center for Arrhythmia Research and Therapeutics, Division of Clinical Pharmacology, Department of Medicine, Vanderbilt University School of Medicine, Nashville, Tennessee, USA.
Matthew J WleklinskiVanderbilt Center for Arrhythmia Research and Therapeutics, Division of Clinical Pharmacology, Department of Medicine, Vanderbilt University School of Medicine, Nashville, Tennessee, USA.
Nieves Gomez-HurtadoVanderbilt Center for Arrhythmia Research and Therapeutics, Division of Clinical Pharmacology, Department of Medicine, Vanderbilt University School of Medicine, Nashville, Tennessee, USA.
Raghav VenkataramanDepartment of Biomedical Engineering, Vanderbilt University, Nashville, Tennessee, USA.
Chelsea E GibbsDepartment of Bioengineering, University of Washington, Seattle, Washington, USA.
Franz J BaudenbacherDepartment of Biomedical Engineering, Vanderbilt University, Nashville, Tennessee, USA.
Shiaoching GongLaboratory of Molecular Biology, Rockefeller University, New York, New York, USA.
Glenn I FishmanLeon H. Charney Division of Cardiology, Department of Medicine, New York University School of Medicine, New York, New York, USA.
Patrick M BoyleDepartment of Bioengineering, University of Washington, Seattle, Washington, USA.
Karl PfeiferDivision of Intramural Research, Eunice Kennedy Shriver National Institute of Child Health and Human Development, NIH, Bethesda, Maryland, USA.
Bjorn C KnollmannVanderbilt Center for Arrhythmia Research and Therapeutics, Division of Clinical Pharmacology, Department of Medicine, Vanderbilt University School of Medicine, Nashville, Tennessee, USA.
Vanderbilt University · USUniversity of Washington · USEunice Kennedy Shriver National Institute of Child Health and Human Development · USNew York University · USRockefeller University · US

Funding

Tumor Immunology and Microenvironment Research ProgramP30CA068485 · NCI · VANDERBILT UNIVERSITY MEDICAL CENTER · PI STACIE B DUSETZINA · 1995 to 2026
$172.8M
Translational Analysis CoreP30DK058404 · NIDDK · VANDERBILT UNIVERSITY MEDICAL CENTER · PI RICHARD M. PEEK · 2002 to 2026
$29.9M
Vanderbilt Diabetes Research CenterP30DK020593 · NIDDK · VANDERBILT UNIVERSITY MEDICAL CENTER · PI ALVIN C POWERS · 2012 to 2026
$29.3M
MEDICAL SCIENTIST TRAINING PROGRAMT32GM007347 · NIGMS · VANDERBILT UNIVERSITY · PI WILLIAMS, CHRISTOPHER S. · 1985 to 2023
$26.3M
Regulated expression and developmental functions of the H19 long noncoding RNAZIAHD001804 · NICHD · EUNICE KENNEDY SHRIVER NATIONAL INSTITUTE OF CHILD HEALTH & HUMAN DEVELOPMENT · PI PFEIFER, KARL ERIC · 2009 to 2025
$20.2M
Shop Module CoreP30EY008126 · NEI · VANDERBILT UNIVERSITY MEDICAL CENTER · PI David J. Calkins · 1989 to 2026
$19.6M
Vanderbilt Mouse Metabolic Physiology CenterU24DK059637 · NIDDK · VANDERBILT UNIVERSITY · PI WASSERMAN, DAVID H · 2001 to 2015
$14.9M
Toward a Mechanism-Based Approach to Treating Cardiac ArrhythmiaR35HL144980 · NHLBI · VANDERBILT UNIVERSITY MEDICAL CENTER · PI KNOLLMANN, BJORN C · 2019 to 2025
$7.1M
Purkinje Cells and Arrhythmia MechanismsR01HL105983 · NHLBI · NEW YORK UNIVERSITY SCHOOL OF MEDICINE · PI FISHMAN, GLENN I · 2011 to 2019
$5.2M
Medical Scientist Training ProgramT32GM152284 · NIGMS · VANDERBILT UNIVERSITY · PI Christopher S. Williams · 2024 to 2026
$4.8M
TRAINING PROGRAM IN ION CHANNEL AND TRANSPORTER BIOLOGYT32NS007491 · NINDS · VANDERBILT UNIVERSITY MEDICAL CENTER · PI DENTON, JEROD S. · 2001 to 2020
$4.1M
Development of a Mirror-Image Natural Product as an Antiarrhythmic TherapeuticR01HL151223 · NHLBI · VANDERBILT UNIVERSITY · PI JOHNSTON, JEFFREY NICHOLAS · 2020 to 2023
$3.1M
NCI NIH HHS P30 CA068485NEI NIH HHS P30 EY008126NHLBI NIH HHS F30 HL145917NHLBI NIH HHS F32 HL140874NHLBI NIH HHS R01 HL105983NHLBI NIH HHS R01 HL146107NHLBI NIH HHS R01 HL151223NHLBI NIH HHS R35 HL144980NIDDK NIH HHS P30 DK020593NIDDK NIH HHS P30 DK058404NIDDK NIH HHS U24 DK059637NIGMS NIH HHS T32 GM007347NIGMS NIH HHS T32 GM152284NINDS NIH HHS T32 NS007491
6 · The paper itself

Abstract

Catecholaminergic polymorphic ventricular tachycardia (CPVT) is an arrhythmia syndrome caused by gene mutations that render RYR2 Ca release channels hyperactive, provoking spontaneous Ca release and delayed afterdepolarizations (DADs). What remains unknown is the cellular source of ventricular arrhythmia triggered by DADs: Purkinje cells in the conduction system or ventricular cardiomyocytes in the working myocardium. To answer this question, we used a genetic approach in mice to knock out cardiac calsequestrin either in Purkinje cells or in ventricular cardiomyocytes. Total loss of calsequestrin in the heart causes a severe CPVT phenotype in mice and humans. We found that loss of calsequestrin only in ventricular myocytes produced a full-blown CPVT phenotype, whereas mice with loss of calsequestrin only in Purkinje cells were comparable to WT mice. Subendocardial chemical ablation or restoration of calsequestrin expression in subendocardial cardiomyocytes neighboring Purkinje cells was sufficient to protect against catecholamine-induced arrhythmias. In silico modeling demonstrated that DADs in ventricular myocardium can trigger full action potentials in the Purkinje fiber, but not vice versa. Hence, ectopic beats in CPVT are likely generated at the Purkinje-myocardial junction via a heretofore unrecognized tissue mechanism, whereby DADs in the ventricular myocardium trigger full action potentials in adjacent Purkinje cells.

Indexed as

Gene Expression RegulationAnimalsCalsequestrinCell LineDisease Models, AnimalHeart RateMiceMice, KnockoutPolymorphic Catecholaminergic Ventricular TachycardiaPurkinje CellsRNATachycardia, VentricularCalsequestrincasq2 protein, mouseRNAArrhythmiasCalcium signalingCardiologyGenetic diseases

Identifiers

PMID34990403
PMCPMC8855823
OpenAlexW4206438053

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Registered trials

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Read under generation 80e0d062 · epoch 390. Bibliography from PubMed, PubMed Central and OpenAlex; grants from NIH RePORTER; trial links from ClinicalTrials.gov; estimates, votes and beliefs from the OpenQuestion graph.