Journal of Arrhythmia 31 (2015) 58–59

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Case Report

A case of cardiac sarcoidosis presenting with double tachycardia Keiko Takahashi, MD, Yasuo Okumura, MDn, Ichiro Watanabe, MD, Koichi Nagashima, MD, Hiroaki Mano, MD, Kazumasa Sonoda, MD, Rikitake Kogawa, MD, Naoko Sasaki, MD, Kazuki Iso, MD, Kimie Okubo, MD, Toshiko Nakai, MD, Atsushi Hirayama, MD Division of Cardiology, Department of Medicine, Nihon University School of Medicine, Tokyo, Japan

art ic l e i nf o

a b s t r a c t

Article history: Received 28 September 2013 Received in revised form 16 February 2014 Accepted 16 May 2014 Available online 27 June 2014

Although the most feared cardiac manifestation in cardiac sarcoidosis is the onset of ventricular arrhythmia, some patients may present with supraventricular arrhythmias. We present a rare case of cardiac sarcoidosis associated with double tachycardia manifesting as atrial flutter and ventricular tachycardia. & 2014 Japanese Heart Rhythm Society. Published by Elsevier B.V. All rights reserved.

Keywords: Cardiac sarcoidosis Double tachycardia Atrial flutter Ventricular arrhythmia

1. Introduction Sarcoidosis is a multisystem granulomatous disease of unknown etiology. In autopsy studies, cardiac involvement has been reported in 40–50% of patients [1]. In cardiac sarcoidosis, ventricular arrhythmias are the most well-known and feared cardiac manifestation, but some of these patients have supraventricular arrhythmias. We present a rare case of cardiac sarcoidosis associated with double tachycardia manifesting as atrial flutter and ventricular tachycardia. 2. Case report The patient was a 60-year-old man who experienced palpitations for the past 6 months. During a medical clinic visit, atrial tachycardia (AT) was suggested on 12-lead electrocardiography (ECG) with a heart rate of 75 beats/min and 3–5:1 atrioventricular conduction. The Holter ECG recorded the AT and frequent ventricular tachycardia (VT) episodes of 3–18 beats (Fig. 1A). Because the AT and VT were drug-refractory, he was referred to our hospital for an electrophysiologic study and catheter ablation. Echocardiography revealed dilatation of the right and left atria and a mildly reduced left ventricular (LV) systolic function with n Correspondence to: 30-1 Ohyaguchi kami-cho, Itabashi-ku, Tokyo 173-8610, Japan. Tel.: þ 81 3 3972 8111; fax: þ 81 3 3972 1098. E-mail address: [email protected] (Y. Okumura).

an aneurysm on the basal posterior LV wall. Blood examination results were normal, including angiotensin converting enzyme levels. Chest radiography and computed tomography (CT) revealed cardiac dilatation, but lung parenchyma lymph nodes were not swollen. Contrast-enhanced magnetic resonance imaging (MRI) revealed delayed enhancement of the aneurysm. The coronary angiogram was normal. The 18F-fluorodeoxyglucose positron emission tomography (PET)–CT revealed extensive hypermetabolic activity in the basal posterior septum and LV posterolateral wall. Together, these findings suggested the presence of cardiac sarcoidosis. A voltage map obtained during the electrophysiologic study revealed low-voltage areas in the high anterior and lateral portions of the right atrium. Atrial activation during the AT occurred in the counterclockwise direction around the tricuspid valve. Pacing from the cavotricuspid isthmus exhibited concealed entrainment; therefore, typical atrial flutter (AFL) was diagnosed. The AFL was terminated by ablation of the cavotricuspid isthmus. Thereafter, sustained monomorphic VT, with the morphology of a right-bundle branch block and superior axis, was induced by programmed right ventricular stimulation. An LV voltage map obtained during sinus rhythm revealed a low-voltage area with delayed potentials in the posterior LV aneurysm. The activation map during VT revealed a focal reentrant pattern with a clockwise rotation in the posterior LV aneurysm, where a mid-diastolic potential was recorded (Fig. 1B). Pacing from this site demonstrated concealed entrainment (Fig. 1C). These findings strongly suggested that the mechanism of the VT was reentry. The VT

http://dx.doi.org/10.1016/j.joa.2014.05.004 1880-4276/& 2014 Japanese Heart Rhythm Society. Published by Elsevier B.V. All rights reserved.

K. Takahashi et al. / Journal of Arrhythmia 31 (2015) 58–59

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Fig. 1. Atrial tachycardia (red arrow) with 3:1 atrioventricular conduction and 18 beats of ventricular tachycardia simultaneously observed on Holter electrocardiography are shown (A). The three-dimensional activation map (inferior view) obtained during ventricular tachycardia (VT) (B). Concealed entrainment observed during VT when pacing was performed at the site of the mid-diastolic potential (DP) (C). The yellow arrows indicate a DP.

terminated 7.8 s after commencing radiofrequency ablation, and was no longer inducible by programmed ventricular pacing. 3. Discussion Ventricular arrhythmias are the most well-known and feared cardiac manifestations of cardiac sarcoidosis. However, AFL is relatively rare, with a reported prevalence of 5% [2]. In this case, MRI and 18F-FDG PET–CT strongly supported a diagnosis of cardiac sarcoidosis, and the patient had typical AFL; however, the morphology showed an atypical AT on ECG. This case of cardiac sarcoidosis was rare, not only because of the supraventricular tachyarrhythmia but also because of the double tachycardias, i.e., simultaneous VT and AFL. The etiology of these tachyarrhythmias remains unknown. Previous cases of cardiac sarcoidosis with ATs were associated with systemic hypertension, diastolic dysfunction, and LA enlargement. Further, atrial fibrillation was the most common clinical manifestation [2]. Their results suggested that ATs could be related to atrial dilatation secondary to LV dysfunction. In fact, arrhythmias may be less commonly caused by direct granulomatous involvement of the atria [3–5]. However, this case had a typical AFL with right-sided dominant atrial dilatation and scar formation, suggesting a different mechanism. For example, noncaseating granulomas may favor involvement of specific regions of the septum and basal ventricle, but eventually become widespread, and may even involve the atria as well. Although a

pathological examination failed to finalize the underlying mechanism of these double tachyarrhythmias, this case highlights the fact that in some cases of cardiac sarcoidosis, the ventricles as well as the atria may be involved in producing arrhythmias of not only a ventricular but also an atrial origin.

Disclosures and funding sources None.

Conflicts of interest None. References [1] Silverman KJ, Hutchins GM, Bulkley BH, et al. Cardiac sarcoid. Circulation 1978;58:1204–11. [2] Viles-Gonzalez JF, Pastori L, Fischer A, et al. Supraventricular arrhythmias in patients with cardiac sarcoidosis: prevalence, predictors, and clinical implications. Chest 2013;143:1085–90. [3] Gozo Jr EG, Cosnow I, Cohen HC, et al. The heart in sarcoidosis. Chest 1971;60:379–88. [4] Wait JL, Movahed A. Anginal chest pain in sarcoidosis. Thorax 1989;44:391–5. [5] Kim JS, Judson MA, Donnino R, et al. Cardiac sarcoidosis. Am Heart J 2009;157:9–21.

A case of cardiac sarcoidosis presenting with double tachycardia.

Although the most feared cardiac manifestation in cardiac sarcoidosis is the onset of ventricular arrhythmia, some patients may present with supravent...
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