Evidence map›Paper›PMID 32301586›Full record

ArticleESC heart failure2020

Monozygotic twins with myocarditis and a novel likely pathogenic desmoplakin gene variant.

Antheia Kissopoulou, Eva Fernlund, Christina Holmgren, Eira Isaksson, Jan-Erik Karlsson, Henrik Green, Jon Jonasson, Rada Ellegård, Hanna Klang Årstrand, Anneli Svensson and 1 more

Open access · goldAbstract readCase Reports
In one paragraph

Article in ESC heart failure, 2020. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 16 papers.

0numbers the graph read from it
0cells of the map it votes in
16citing papers in PubMed
2.5field-weighted citation impact, top 9% 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

16 citing papers in PubMed, 23 citations in OpenAlex.

  1. Genotype-phenotype correlations inFrontiers in cardiovascular medicine · 2026
    Article
  2. Article
  3. Article
  4. Red Flags in Acute Myocarditis.Cardiac failure review · 2024
    Review
  5. Article
  6. Article
  7. Article
  8. Frontiers in cardiovascular medicine · 2023
    Article
  9. Review
  10. Article
  11. Article
  12. Review
  13. Myopathic Cardiac Genotypes Increase Risk for Myocarditis.JACC. Basic to translational science · 2021
    Article
  14. Review
  15. A NovelDiagnostics (Basel, Switzerland) · 2020
    Article
  16. 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

11 authors at 4 institutions in 1 country.

Antheia KissopoulouDepartment of Internal Medicine, County Council of Jönköping, Jönköping, Sweden.
Eva FernlundCrown Princess Victoria Children's Hospital, Division of Pediatrics, Department of Biomedical and Clinical Sciences, Linköping University, Linköping University Hospital, Linköping, Sweden.
Christina HolmgrenDepartment of Internal Medicine, County Council of Jönköping, Jönköping, Sweden.
Eira IsakssonDepartment of Internal Medicine, County Council of Jönköping, Jönköping, Sweden.
Jan-Erik KarlssonDepartment of Internal Medicine, County Council of Jönköping, Jönköping, Sweden.
Henrik GreenDivision of Drug Research, Department of Health, Medicine and Caring Sciences, Linköping University, Linköping, Sweden.
Jon JonassonDepartment of Clinical Genetics and Department of Biomedical and Clinical Sciences, Linköping University, Linköping, Sweden.
Rada EllegårdDepartment of Clinical Genetics and Department of Biomedical and Clinical Sciences, Linköping University, Linköping, Sweden.
Hanna Klang ÅrstrandDepartment of Clinical Genetics and Department of Biomedical and Clinical Sciences, Linköping University, Linköping, Sweden.
Anneli SvenssonDepartment of Cardiology, Department of Health, Medicine and Caring Sciences, Linköping University, Linköping, Sweden.
Cecilia GunnarssonDepartment of Clinical Genetics and Department of Biomedical and Clinical Sciences, Linköping University, Linköping, Sweden.
Linköping University · SELinköping University Hospital · SERegion Jönköpings län · SESwedish National Board of Forensic Medicine · SE

Funding

FORSSFuturumRegion Östergötland
6 · The paper itself

Abstract

Myocarditis most often affects otherwise healthy athletes and is one of the leading causes of sudden death in children and young adults. Arrhythmogenic right ventricular cardiomyopathy (ARVC) is a genetically determined heart muscle disorder with increased risk for paroxysmal ventricular arrhythmias and sudden cardiac death. The clinical picture of myocarditis and ARVC may overlap during the early stages of cardiomyopathy, which may lead to misdiagnosis. In the literature, we found several cases that presented with episodes of myocarditis and ended up with a diagnosis of arrhythmogenic cardiomyopathy, mostly of the left predominant type. The aim of this case presentation is to shed light upon a possible link between myocarditis, a desmoplakin (DSP) gene variant, and ARVC by describing a case of male monozygotic twins who presented with symptoms and signs of myocarditis at 17 and 18 years of age, respectively. One of them also had a recurrent episode of myocarditis. The twins and their family were extensively examined including electrocardiograms (ECG), biochemistry, multimodal cardiac imaging, myocardial biopsy, genetic analysis, repeated cardiac magnetic resonance (CMR) and echocardiography over time. Both twins presented with chest pain, ECG with slight ST-T elevation, and increased troponin T levels. CMR demonstrated an affected left ventricle with comprehensive inflammatory, subepicardial changes consistent with myocarditis. The right ventricle did not appear to have any abnormalities. Genotype analysis revealed a nonsense heterozygous variant in the desmoplakin (DSP) gene [NM_004415.2:c.2521_2522del (p.Gln841Aspfs*9)] that is considered likely pathogenic and presumably ARVC related. There was no previous family history of heart disease. There might be a common pathophysiology of ARVC, associated with desmosomal dysfunction, and myocarditis. In our case, both twins have an affected left ventricle without any right ventricular involvement, and they are carriers of a novel DSP variant that is likely associated with ARVC. The extensive inflammation of the LV that was apparent in the CMR may or may not be the primary event of ARVC. Nevertheless, our data suggest that irrespective of a possible link here to ARVC, genetic testing for arrhythmogenic cardiomyopathy might be advisable for patients with recurrent myocarditis associated with a family history of myocarditis.

Indexed as

Arrhythmogenic Right Ventricular DysplasiaMyocarditisAdolescentDesmoplakinsGenetic TestingHumansMaleTwins, MonozygoticDesmoplakinsArrhytmogenic cardiomyopathyDesmoplakin geneMyocarditis

Identifiers

PMID32301586
PMCPMC7261567
OpenAlexW3016600641

What OpenQuestion holds

Textmetadata
LicenceCC BY-NC
Read underepoch 390

Registered trials

None linked

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.