Abstract
A 47-year-old woman with Rosai–Dorfman disease (RDD) presented with an electrical
storm with sustained ventricular tachycardia. Echocardiography and computed tomography
revealed an intracardiac mass involving the right atrium and left ventricle. Positron
emission tomography demonstrated fluorodeoxyglucose avidity of the mass and myocardial
infiltration; the electrical storm was attributed to this feature. Cardiac involvement
in RDD is extraordinarily rare, and this appears to be the first report of an association
of this disease with an electrical storm.
Résumé
Une femme de 47 ans atteinte de la maladie de Rosai-Dorfman (MRD) a présenté un orage
rythmique avec tachycardie ventriculaire soutenue. Les examens par échocardiographie
et par tomodensitométrie ont révélé une masse intracardiaque touchant l’oreillette
droite et le ventricule gauche. La tomographie par émission de positrons a mis au
jour l’avidité de cette masse pour le fluorodésoxyglucose ainsi qu’une infiltration
du myocarde, laquelle expliquerait l’orage rythmique. Il est extrêmement rare que
la MRD touche le cœur, et il semble qu’il s’agisse du premier cas d’orage rythmique
signalé en lien avec cette maladie.
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References
- Consensus recommendations for the diagnosis and clinical management of Rosai–Dorfman–Destombes disease.Blood. 2018; 131: 2877-2890
- Rosai–Dorfman disease and the heart.Intractable Rare Dis Res. 2016; 5: 1-5
- Isolated cardiac involvement of Rosai-Dorfman disease.Ann Thorac Surg. 2012; 94: 2118-2120
- Editor’s Choice–The treatment of electrical storm: an educational review.Eur Heart J Acute Cardiovasc Care. 2018; 7: 478-483
- Sinus histiocytosis with massive lymphadenopathy: extra-nodal Rosai-Dorfman disease presenting as a rare aetiology of a large intracardiac mass.Eur Heart J. 2017; 38: 1439-1440
Article info
Publication history
Published online: March 06, 2020
Accepted:
March 4,
2020
Received:
October 18,
2019
Footnotes
See page 1326.e15 for disclosure information.
Identification
Copyright
© 2020 Canadian Cardiovascular Society. Published by Elsevier Inc. All rights reserved.
