P.O. Box 2925 Riyadh – 11461KSATel: +966 1 2520088 ext 40151Fax: +966 1 2520718Email: sha@sha.org.saURL: www.sha.org.sa

CA

SE R

EPO

RTDisclosure: Authors have nothing to disclose with regard tocommercial support.

Received 26 July 2013; accepted 2 August 2013.Available online 13 August 2013

⇑ Corresponding author. Tel.: +216 98 632 680.E-mail addresses: zied.bh@laposte.net (Z. Ibn Elhadj), salem.kachboura@

rns.tn (S. Kachboura).

Cardiac hydatid cyst revealed by ventriculartachycardia

1016–7315 � 2013 Production and hosting by Elsevier B.V. on behalf of King Saud University.

Peer review under responsibility of King Saud University.

URL: www.ksu.edu.sa

http://dx.doi.org/10.1016/j.jsha.2013.08.001 Production and hosting by Elsevier

Zied Ibn Elhadj a,b,⇑, Marouane Boukhris a,b, Ikram Kammoun a,b, Afef Ben Halima a,b,Faouzi Addad a,b, Salem Kachboura a,b

a Service de Cardiologie, Hôpital Abderrahmen Mami, Tunis El Manar, 2080 Ariana; b Faculty of Medecine, Tunis El Manar;a,b Tunisia

Hydatid disease is a human parasitic infestation caused by the larval stage of Echinococcus Granulosus. The liver andthe lungs are the most common locations. Cardiac involvement is rare and accounts for 0.5–2% of all hydatid disease.We report an unusual presentation of cardiac hydatid cyst revealed by ventricular tachycardia in a patient with a historyof cerebral hydatid cyst.

� 2013 Production and hosting by Elsevier B.V. on behalf of King Saud University.

Keywords: Hydatid disease, Cardiac hydatid cyst, Ventricular tachycardia

Introduction

Hydatid disease is a human parasitic infesta-tion caused by the larval stage of Echinococ-

cus Granulosus. The liver and the lungs are themost common locations. Cardiac involvement israre and accounts for 0.5–2% of all hydatid disease[1]. Solitary cardiac cysts can be asymptomaticover long periods, or can be discovered after seri-ous and even fatal complications. Rupture re-mains the major life threatening complicationand may result in anaphylactic shock. Cardiactamponade, systemic or pulmonary embolism[2], chest pain [3], valvular regurgitation [4], and– in exceptional cases – arrhythmias can revealhydatid cysts.[5]

Case report

A 24-year-old man was admitted to our inten-sive care unit for chest pain and palpitations. Hehad been operated on when he was nine yearsold for a brain hydatid cyst of the right temporo-parietal area.

On physical examination, heart rate was200 bpm, blood pressure was 100/60 mmHg, andthere were no signs of heart failure. Neurologicalexamination was normal. Electrocardiogram(ECG) showed a ventricular tachycardia (VT) witha heart rate of 200 bpm (Fig. 1). Sinus rhythm wassuccessfully restored after intravenous infusion ofamiodarone. No cardiac or pulmonary abnormali-ties were found on chest X-ray. Trans-thoracic

CA

SE REPO

RT

Figure 1. ECG: ventricular tachycardia with a heart rate of 200 bpm.

48 IBN ELHADJ ET ALCARDIAC HYDATID CYST

J Saudi Heart Assoc2014;26:47–50

echocardiography revealed the presence of a het-erogeneous intramyocardial tumor with intratu-moral calcifications, covering two thirds of theapico-lateral left ventricular wall (Fig. 2). Cardiacmagnetic resonance imaging (MRI) showed a het-erogeneous well-defined intramyocardial tumordisplaying hyperintensity on T1 and T2 weightedMRI (Fig. 3a and b). Cardiac computed tomogra-phy (CT) was performed and found a large intra-myocardial cystic mass measuring 5.8 � 5 cminvolving the apico-lateral left ventricular wallwith well-defined partially calcified limits and noenhancement after contrast injection (Fig. 4).Enzyme linked immunosorbent assay (ELISA) test

for Echinococcus was negative. Laboratory testswere normal. The diagnosis of cardiac hydatidcyst was made and a surgical removal wasindicated.

On intervention, an abnormal convexity of theanterolateral and apical wall of the left ventriclewas found. The cyst was punctured and rinsedwith hypertonic saline solution to destroy daugh-ter cysts, after which total cystectomy was per-formed except for the pericyst tissue thatadhered to the left ventricular wall. Histologicexamination confirmed the diagnosis of a hydatidcyst. The postoperative period was uneventful,and the patient was discharged home on the tenth

CA

SE R

EPO

RT

Figure 3. (a and b) Cardiac magnetic resonance imaging showing aheterogeneous well-defined intramyocardial tumor displaying hyperintensity on T1 and T2 weighted MRI.

Figure 4. Cardiac computed tomography showing a large intramyocardial cystic mass (5.8 � 5 cm) involving the apico-lateral leftventricular wall with well-defined partially calcified edges.

Figure 2. Trans-thoracic echocardiography showing a heterogeneousintramyocardial tumor with intra tumoral calcifications, covering twothirds of the apico-lateral left ventricular wall.

J Saudi Heart Assoc2014;26:47–50

IBN ELHADJ ET AL 49CARDIAC HYDATID CYST

day under amiodarone. There was no recurrenceof ventricular tachycardia and amiodarone wasstopped three months later. Control trans-thoracicechocardiography showed a residual cavity of4 cm2. Five years later, the patient is still doingwell with no symptoms.

Comment

Since the first report of cardiac hydatidosis byWilliams in 1836, cases of cardiac hydatid diseasehave increasingly been reported [6]. Hydatid dis-ease is endemic in tropical and subtropical re-gions, particularly in the Mediterranean basin,South America, Africa and Australia [1]. Cardiacinvolvement is rare [1] and often asymptomatic.Only 10% of patients, especially those with largehydatid cysts have clinical manifestations.

In most cases, a vague chest pain or typical an-gina had been reported and may be secondaryto coronary arteries compression. Myocardialinfarction is exceptional [5]. Pericardial type painis encountered when the pericardium is involved.The cysts are commonly located over the interven-tricular septum and can be responsible for varyingdegrees of atrioventricular and intraventricularblocks [8]. The cyst location may induce obstruc-tion of right [9] or left ventricular outflow tracts[10]. When the cyst is located near the valvularapparatus, it may simulate valvular stenosis orcause valvular regurgitation [4,11]. Rupture re-mains the major life threatening complicationand may result in anaphylactic shock, cardiactamponade and systemic or pulmonary embolism

CA

SE REPO

RT

50 IBN ELHADJ ET ALCARDIAC HYDATID CYST

J Saudi Heart Assoc2014;26:47–50

[2]. Cardiac arrhythmias are exceptional and tothe best of our knowledge, ventricular tachycardiawas reported in less than 20 cases [5]. The mecha-nism of the VT is uncertain. VT reproductivity byventricular programmed stimulation in othercases reported in literature [12], suggests a re-entry mechanism. The absence of recurrence ofthe VT after surgical removal of the cyst impliesalteration of automatic substrate for VT by theresection, even if a postoperative residual cavitywas noticed. It suggests that the cyst surroundingcardiac fibers may represent the anatomic sub-stratum for the VT.

There are no specific abnormalities for cardiachydatid cyst on physical examination and ECG.Hypereosinophilia is not specific either and israrely encountered. The presence of high titersof anti-Ecchinococcus antibodies is helpful but anegative serology, as in our patient, cannot ruleout the diagnosis [13]. Two-dimensional echocar-diography is the best tool for the detection andlocalization of cardiac involvement. A trans-thoracic echocardiography should therefore beperformed when a non- cardiac hydatid diseaseis diagnosed. The cardiac hydatid cyst is often sin-gle with well-defined edges and sometimes withinternal trabeculae corresponding to daughtervesicles [1–7]. CT scan and MRI are helpful whenthe hydatid cyst presents as solid mass due todegenerative process. They also allow the diagno-sis of other thoracic cysts that need to be treated atthe same time [14]. Coronary angiography is indi-cated in case of angina, in elderly patients andwhen the cyst is adjacent to coronary arteries [6].As the outcome is unpredictable, surgical removalremains the best treatment, even in asymptomaticpatients, because of the risk of rupture [2]. Data onthe medical treatment of cardiac hydatid diseaseare limited. Albendazole was reported to be suc-cessful in some sporadic cases [15]. Some authorshave reported on the association of cerebral andcardiac hydatid cysts which are generally discov-ered at the same time [15]. The brain cyst maybe due to the rupture of the cardiac cyst with mul-tiple cerebral hydatid embolization [15].

Liver and lungs are still the most common loca-tions. The originality of our case is the long periodseparating the discovery of these two locationswith absence of liver or lung involvement. Twohypotheses are possible: the presence of a verysmall cardiac cyst which was not detectable byechocardiography at the time of cerebral cystdiscovery and re-infestation with EchinococcusGranulosus. The final question remains: are thesetwo cysts due to the same infestation?

Conclusion

Cardiac hydatid cyst is rare but may be lifethreatening. It is often asymptomatic but can berevealed by arrhythmias. Cardiac involvementcan be isolated or associated with other locations.Therefore, an echocardiography should be alwaysperformed in patients presenting with non-cardiac hydatid disease.

References

[1] Ameli M, Mobarhan HA, Nouraii SS. Surgical treatment ofhydatid cyst of the heart: report of six cases. J ThoracCardiovasc Surg 1989;98(5 Pt 2):892–901.

[2] Perez-Gomez F, Duran H, Tamames S, Perrote JL, BlanesA. Cardiac echinococcosis: clinical picture andcomplications. Br Heart J 1973;35(12):1326–31.

[3] Kosar F, Aksoy Y, Sahin I, Erdil N. Pericardial hydatid cystmimicking acute coronary syndrome. Tex Heart Inst J2005;32(4):570–2.

[4] Apaydin AZ, Oguz E, Ayik F, Nalbantgil S, Ceylan N.Hydatid cyst confined to the papillary muscle: a very rarecause of mitral regurgitation. Tex Heart Inst J.2009;36(6):598–600.

[5] Sirinelli A, Le Guludec D, Laine JF, Sebag C, BourguignonM, Slama M, et al. Ventricular tachycardia revealing ahydatid cyst. Am Heart J 1987;114(3):656–9.

[6] Yekeler I, Koçak H, Aydin NE, Bas�oglu A, Okur A, SenocakH, et al. A case of cardiac hydatid cyst localized in thelungs bilaterally and on anterior wall of right ventricle.Thorac Cardiovasc Surg 1993;41(4):261–3.

[7] Elangovan S, Harshavardan K, Meenakshi K,Swaminathan TS, Murthy P. Left ventricular hydatid cystwith myocardial infarction in a patient with severerheumatic mitral stenosis. Indian Heart J 2004;56(6):664–7.

[8] De Los Arcos E, Madurga MP, Perez Leon J, Martinez JL,Urquia M. Hydatid cyst of the interventricular septumcausing left anterior hemiblock. Br Heart J 1971;33(4):623–5.

[9] Oliver JM, Sotillo JF, Domínguez FJ, López de Sá E, CalvoL, Salvador A, et al. Two-dimensional echocardiographicfeatures of echinococcosis of the heart and great bloodvessels. Clinical and surgical implications. Circulation1988;78(2):327–37.

[10] Lanzoni AM, Barrios V, Moya JL, Epeldegui A, Celemin D,Lafuente C, et al. Dynamic left ventricular outflowobstruction caused by cardiac echinococcosis. Am HeartJ 1992;124(4):1083–5.

[11] Gomez FP, Duran H, Tamames S, Perrote JL, Blames A.Cardiac echinococcosis: clinical picture and complications.Br Heart J 1973;35:1326–31.

[12] Akar R, Eryilmaz S, Yazicioglu L, Eren NT, Durdu S, UysalelA, et al. Surgery of cardiac hydatid disease: an Anatolianexperience. Anadolu Kardiyol Derg 2003;3(3):238–44.

[13] Ben-Hamda K, Maatouk F, Ben-Farhat M, Betbout F,Gamra H, Addad F, et al. Eighteen-year experience withechinococcosis of the heart: clinical and echocardiographicfeatures in 14 patients. Int J Cardiol 2003;91(2–3):145–51.

[14] Koubâa M, Lahiani D, Abid L, Mâaloul I, Kahla SB, BradiiM, et al. Can albendazole be the only treatment for cardiacechinococcosis with multiple organ involvement? Int JCardiol 2012;161(3):58–60.

[15] Bükte Y, Kemaloglu S, Nazaroglu H, Ozkan U, Ceviz A,Simsek M. Cerebral hydatid disease: CT and MR imagingfindings. Swiss Med Wkly 2004;134(31–32):459–67.