International Journal of Cardiology 176 (2014) 1192–1194

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Letter to the Editor

Acute eosinophilic myocarditis Xianda Ni a,1, Jing Ping Sun b,1, Xing Sheng Yang b, Cheuk-man Yu b,⁎ a b

Department of Ultrasonography, The First Affiliated Hospital of Wenzhou Medical University, Wenzhou, Zhejiang, China Prince of Wales Hospital, Chinese University of Hong Kong, Hong Kong, China

a r t i c l e

i n f o

Article history: Received 11 July 2014 Accepted 27 July 2014 Available online 4 August 2014 Keywords: Acute myocarditis Echocardiography MRI

A 43-year-old man was admitted to our hospital with fever, chills, persistent headache accompanied by dizziness, nausea, vomiting. He had a history of hypertension 4 years; gout 5 years. Physical examination showed cervical resistance and Klinefelter syndrome (+). Laboratory tests revealed elevated levels of white blood neutrophils, eosinophil cell count, cardiac troponins and C-reactive protein. Cerebrospinal fluid: leukocytosis, Pan's test (+), the level of protein was increased, while the level of glucose and chloride was decreased. Electrocardiogram (ECG): sinus rhythm, normal. Magnetic resonance imaging (MRI): revealed multiple lesions scattered in the brain, the possibility of tuberculous meningitis was considered. The antituberculous meningitis therapy was begun. One week later, patient felt severe chest pain. ECG: showed ST-T change in all leads: bone marrow hyperplasia, myeloid hyperplasia, increased eosinophils, giant hyperplasia. All myocardial enzymes, troponin and C-reactive protein were significantly elevated. Because an electrocardiogram showed new ST-segment elevation, he was taken for emergency coronary angiography, which revealed normal coronary artery. A computed tomographic scan of the chest revealed both sides of the pleural effusion, and cardiac shadow enlarged with pericardial effusion. Transthoracic echocardiography revealed thickness of left ventricular walls and right ventricular free wall was significantly increased, echogenic myocardium with normal ejection fraction

⁎ Corresponding author at: Division of Cardiology, Department of Medicine and Therapeutics, Prince of Wales Hospital, Li Ka Shing Institute of Health and Sciences, The Chinese University of Hong Kong, Hong Kong, China. Tel.: +852 2632 3594; fax: +852 2637 5643. E-mail address: [email protected] (C. Yu). 1 Co-first author.

http://dx.doi.org/10.1016/j.ijcard.2014.07.233 0167-5273/© 2014 Elsevier Ireland Ltd. All rights reserved.

(Fig. 1), left ventricular diastolic function was impaired (E/E′ = 22), moderate mitral regurgitation, moderate to severe tricuspid regurgitation and mild pulmonary hypertension. The characteristics of echocardiography suggested an infiltrative disorder. MRI scans showed abnormal delayed enhancement signal within the myocardium, pericardial effusion; (Fig. 2. A,B,C) and abnormal contrast enhancement in myocardium (Fig. 2. C,D,E). Endomyocardial biopsy: Microscopically, myocardial disarray, eosinophilic infiltrate can be seen in myocardial interstitial focal area of myocardial endoplasmic (arrows), in line with addicted eosinophil endocarditis and myocarditis (Fig. 3). The cardiac arrest occurred suddenly, and he fortunately recovered by cardiopulmonary resuscitation during examinations. According to the characteristics of echocardiography, MRI and endomyocardial biopsy, the diagnosis of eosinophilic myocarditis was confirmed. The patient received prednisone combined with anti-tuberculosis therapy. A week later, the symptoms of the patient had improved, and the count of eosinophils significantly decreased. After three months, chocardiography showed: the thickened myocardium of left and right ventricle was significantly improved, atrioventricular valve regurgitation significantly reduced, pericardial effusion disappeared and E/E′ returned to 8. MRI showed the lesions in myocardium apparently improved. Discussion: Eosinophil endocarditis and myocarditis most likely occurred as a result of hypersensitivity reaction. Drugs typically associated with the occurrence of eosinophilic myocarditis [1,2]. Patients with eosinophilic myocarditis may present with various signs and symptoms including fever, chills, malaise, weight loss, acute coronary syndromelike features, acute coronary syndrome-like features, heart failure, tachy- or brady-type arrhythmias, and sudden death [3,4]. To date there are no universally accepted guidelines for the diagnosis of eosinophilic myocarditis. Essential diagnostic features include eosinophilia N500/μL, cardiac symptoms, elevated cardiac enzymes, electrocardiogram (ECG) changes, and cardiac dysfunction on ultrasonography, especially in the setting of unremarkable coronary angiography. Definitive diagnosis requires an endomyocardial biopsy [5]. In the present case, the initial disease was tuberculous meningitis, anti-tuberculous meningitis drugs were involved. One week after anti-tuberculous therapy, patient felt severe chest pain. All results of laboratory examinations fulfilled the criteria for a diagnosis of eosinophilic myocarditis, which was confirmed by endomyocardial biopsy. The etiology of eosinophilic myocarditis in this patient might be the anti-tuberculous drugs, but it is hard to exclude the tuberculous itself. Steroids are the cornerstone of treatment in eosinophilic myocarditis [6]. Our patient was given steroid therapy, which was followed by a dramatic response within weeks, with solid

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Fig. 1. Parasternal long axis (A), short axis (B) and apical 4-chamber views revealed thickness of left ventricular walls and right ventricular free wall was significantly increased with echogenic endocardium.

documentation via serial echocardiograms, MRI and biomarker measurements of the course of cardiac dysfunction and recovery. This case also clearly highlights the persistently important role of echocardiography in the evaluation of patients with myocarditis, despite the very prominent role of MRI in this field [7]. It remains indisputable that MRI with late gadolinium enhancement images and edema sequences (as direct evidence of myocardial infiltration and inflammation) provide an excellent tool for the diagnosis of myocarditis [7], particularly if the echocardiogram is inconclusive. But the role of MRI in the evaluation of cardiac function is limited to the accurate measurement of left and right ventricular volumes and ejection fractions. In contrast, transthoracic echocardiography using tissue-Doppler echocardiography enabled excellent functional evaluation at the initial examination and during follow-up in our patient. Ideally, we also have obtained a MRI of our patient at initial and follow-up examinations, the results of which were consistent with the echocardiographic findings; which supported the diagnosis and recovery course solidly. It is important to note that echocardiography, in comparison with MRI, is relatively inexpensive and broadly available. Tissue-Doppler readings can be obtained for nearly all patients even if 2-D quality is imperfect; and experience renders the measurements highly reproducible [8]. In patients with myocarditis, echocardiography clearly cannot replace MRI in general. However, in

patients with obvious abnormalities in the initial echocardiogram, comprehensive echocardiography that includes tissue-Doppler

Fig. 3. Endomyocardial biopsy: Microscopically, myocardial disarray, eosinophilic infiltrate can be seen in myocardial interstitial focal area of myocardial endoplasmic reticulum (see eosinophil infiltration (arrows)), in line with addicted eosinophil endocarditis and myocarditis.

Fig. 2. Magmatic resonance imaging showed abnormal delayed enhancement signal within the myocardium, pericardial effusion; (Fig. 2. A,B,C) and abnormal contrast enhancement in myocardium (Fig. 2. C,D,E).

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measurement can be used for the serial evaluation of LV structure and function during follow-up. Conclusion: Eosinophilic myocarditis remains a rare and likely under-diagnosed subtype of myocarditis. The main characteristics of this disease include myocardial injury in the setting of noncontributory coronary artery disease. Endomyocardial biopsy remains the definitive gold standard for diagnosis of noninfectious myocarditis [9]. Peripheral eosinophilia may strongly suggests the potential for early diagnosis. Missing the early diagnosis of myocarditis and the delay of therapy may lead to irreversible myocardial injury. Large prospective studies in validating therapies of eosinophilic myocarditis are needed. Consent Written informed consent was obtained from patient for publication of this case report and any accompanying images. Copies of the written consent are available for review by the Editor-in-Chief of this journal. Conflict of interest The authors report no relationships that could be construed as a conflict of interest.

References [1] Ginsberg F, Parrillo JE. Eosinophilic myocarditis. Heart Fail Clin 2005;1(3):419–29. [2] Sabatine MS, Poh KK, Mega JL, Shepard JA, Stone JR, Frosch MP. Case records of the Massachusetts General Hospital. Case 36-2007. A 31-year-old woman with rash, fever, and hypotension. N Engl J Med 2007;357(21):2167–78. [3] Al Ali AM, Straatman LP, Allard MF, Ignaszewski AP. Eosinophilic myocarditis: case series and review of literature. Can J Cardiol 2006;22(14):1233–7. [4] Bleakley C, McEneaney D. Eosinophilic myocarditis presenting as acute myocardial infarction. J Cardiovasc Med 2010;11 [000–000]. [5] Talierco CP, Olney BA, Lie JT. Myocarditis related to drug hypersensitivity. Mayo Clin Proc 1985;60:463–8. [6] Amini R, Nielsen C. Eosinophilic myocarditis mimicking acute coronary syndrome secondary to idiopathic hypereosinophilic syndrome: a case report. J Med Case Reports 2010;4:40. [7] Friedrich MG, Sechtem U, Schulz-Menger J, et al. Cardiovascular magnetic resonance in myocarditis: a JACC white paper. J Am Coll Cardiol 2009;53(17):1475–87. [8] Nagueh SF, Appleton CP, Gillebert TC, et al. Recommendations for the evaluation of left ventricular diastolic function by echocardiography. J Am Soc Echocardiogr 2009; 22(2):107–33. [9] Caforio A, Pankuweit S, Arbustini E, et al. Current state of knowledge on aetiology, diagnosis, management, and therapy of myocarditis: a position statement of the European Society of Cardiology Working Group on Myocardial and Pericardial Diseases. Eur Heart J 2013;34(33):2636–48.

Acute eosinophilic myocarditis.

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