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Journal-Cardiovascular Surgery 2013:1(1);13-15 doi; 10.5455/jcvs.2013114 Case Report 13 A rare localization of hydatid cyst: Right ventricular free wall cyst fistulized to the ventricular cavity Tuğra Gençpınar1, Mehmet Güzeloğlu*2, Koray Aykut2, Gökhan Albayrak2, Eyüp Hazan1, Department of Cardiovascular Surgery, State Hospital, Antalya Department of Cardiovascular Surgery, Medical Park Hospital, Izmir University, Turkey * Corresponding Author: Department of Cardiovascular Surgery, Medical Park Hospital, Izmir University Yeni Girne Bulvari Karsiyaka,Izmir/Turkey. Tel: +90 532 7420220 Fax:+90 232 3995070 e-mail: [email protected] Key words: Hydatid Cyst; heart ventricle, right; pulmonary embolism Received: 14.01.2013 Accepted: 21.01.2013 e-published: 15.02.2013 1 2 Abstract Hydatid disease is the result of a parasitic infestation of the larva of ecchinocccus granulosus. Cardiac cyst is a rare complication of the infection. Most frequently, the cyst is localized within the free wall of the left ventricle and interventricular septum, where the blood supply is ample. Pericardium, right ventricle and interatrial septum are rarely affected. Symptoms depend on localization of the cyst. Here, we report a female patient with multiple cysts adherent to right ventricle wall were noted. The patient was successfully treated with surgical therapy. Introduction Hydatic disease is the result of a parasitic infestation of the larva of ecchinocccus granulosus. Cardiac cyst is a rare complication of the infection and it makes up to 0.05-0.2 percent of all cyst hydatid cases. The cyst is frequently localized within the free wall of the left ventricle and interventricular septum, where the blood supply is ample. Pericardium, right ventricle and interatrial septum are rarely affected (1). Symptoms depend on localization of the cyst and the clinical status of the patient. Here we present a young female patient with multiple cysts. Case Report A 29-year-old housewife was referred to our hospital. She was diagnosed with Behçet’s disease, in another institution to which she was presented with cough, shortness of breath, fever and chills. She had a history of an operation for liver hydatid disease 5 years ago. In physical examination, dyspnea and bi-basilar rhales were evident. Chest X-ray revealed parahilar opacity (Figure 1), her electrocardiographic exam was non-specific. We performed an echocardiography. Pulmonary artery systolic pressure was measured as 70mmhg, also multiple cysts adherent to right ventricle wall were noted. A multiloculated cyst measuring 45X36X27 mm in diameter was seen on the inferior wall of the right ventricle in cardiac A rare localization of hydatid cyst: Right ventricular free wall cyst fistulized to the ventricular cavity magnetic resonance imaging (MRI). That particular cyst seemed to be originating from right ventricular epicardium and was extending into the pericardial space. Another smaller 1 cm cyst was also noted which was localized in the endocardium and prolapsing into the right ventricle (Figure 2). Echinococcus antibody was positive at 1/1250 titration with ELISA, erhytrocyte sedimentation rate and C-reactive protein levels were within normal limits. We performed a median sternotomy and administered NO perioperatively. We stopped NO inhalation right after bicaval cannulation and aortic root cold cardioplegia was secured. Within the pericardial space, a cyst approximately of 5 cm in diameter next to right coronary artery was observed (Figure 3a). We performed a cystectomy after securing the area with 3% NaCl soaked gauze (Figure 3b). Exploration revealed that the cyst had already fistulized into the right ventricle. The fistula was repaired with a Gore Tex mesh (Figure 3c). We performed a right ventriculotomy and excised the cyst with careful protection of the papillary muscle (Figure 3d). The right ventricle was washed many times with 3% NaCl solution (Figure 3e). The surgery was completed within 31 minutes of aortic cross clamp and 41 minutes of cardiac bypass time (Figure 3f). We administered post-operative albendazole treatment and scheduled follow up with computerized tomography (CT) angiography and transthoracic echocardiography. Gençpınar et al. Journal-Cardiovascular Surgery 2013:1;13-15 doi; 10.5455/jcvs.2013114 Case Report 14 Figure 2. Hydatid cyst on the inferior wall of the right ventricle detected in cardiac MRI. Figure1. Chest X-ray of the patient. Discussion Hydatid disease is endemic in Mediterranean region, middle east, south Africa and Australia. Embryonated echinococcus eggs are excreted in feces of cats, dogs etc. and humans and are accidentally infested by ingesting contaminated food. Most of those eggs are eliminated by host defenses, those which escape, progress to the cyst stage. Cysts grow slowly, usually at a rate of 1 cm/year (2). When cardiac infection is the case, cysts are encased in an adventitial pericystic coat and they reach maturity in 1-5 years (2). The first cardiac cyst case has been reported by Williams et al. in 1936. Ten years after that paper, Greisinger et al. published a case series of 15 patients (3). Cardiac hydatid infection most commonly affects the left ventricle (%55-60) where the blood supply is ample (4). Interventricular septum, right atrial wall, right ventricle, left atrium, pulmonary artery and pericardium are less commonly reported. Symptoms depend on cyst localization and clinical status of the patient. Cardiac cyst hydatic frequently presents with pericardiac chest pain, dyspnea, cough and fever (1,2). Anaphylaxis and shock may rarely occur. Clinical picture may also include cardiac tamponade, arrhthymias, myocardial infarction, pericarditis, valvular dysfunction, pulmonary and systemic emboli, congestive heart failure and sudden death. Our patient had fever, chills, dyspnea and cough. In diagnosis, most patients exhibit multivisceral involvement (%55-85) (5). Murmurs can be heard. Electrocardiographic findings are not specific to cardiac cyst hydatid. In chest X-ray, calcified lobular masses and abnormal heart shadows can be seen. Echocardiography is the most effective examination in terms of providing a diagnosis and formulat- A rare localization of hydatid cyst: Right ventricular free wall cyst fistulized to the ventricular cavity ing a plan. Tomography scans and MRI can show extracardiac involvement, if present (2). Serologic tests are positive in 50% of the patients and false negativity must be taken into account. We made the diagnosis with echocardiography and soon MRI, and serologic tests were strongly positive. Casoni skin test and Weinberg complement fixation tests are outdated because of low sensitivity and specificity. In terms of medical treatment, benzimidazole derivatives are effective. They inhibit parasite spesific fumarate reductase, microtubule polymerase, oxidative phosphorylation and glucose transport. Because of the risk of rupture into cardiac chambers and pericardium, and sudden death, the mainstay of the cardiac cyst treatment is urgent surgery. In cardiac cyst hydatid, operative mortality is reported as 0,25-0,1% (6). In conclusion, in myocardial involvement of hydatic disease, surgical intervention is preferred because of the risk of sudden death and possible complications. The relative low mortality and morbidity and high effectiveness of the surgery are also suggestive for surgical treatment. Surgical intervention is most commonly performed on-pump. Hydatid disease is still common in our country and a variety of cases such as this case can be encountered in clinical practice. Acknowledgements None Conflicts of Interest The authors declared no conflicts of interest with respect to the authorship and/or publication of this article. Gençpınar et al. Journal-Cardiovascular Surgery 2013:1;13-15 doi; 10.5455/jcvs.2013114 Case Report 15 Figure 3. Intraoperative view of the hydatid cyst. References 1)Akar R, Eryilmaz S, Yazicioglu I, et al. Surgery for cardiac disease: An Anatolian Experience. Anadolu Kardiyol Derg 2003;3:238-244. 2)Kaplan M, Demirtas M, Cimen S, Ozler A. Cardiac hydatid cysts with intracavitary expansion. Ann Thorac Surg 2001;71:1587-1590. 3) Atilgan D, Kudat H, Tukek T, et al. Role of transesophageal echocardiography in diagnosis and management of cardiac hydatid cyst: report of three cases and review of the literature. J Am Soc Echocardiogr 2002;15:271-274. A rare localization of hydatid cyst: Right ventricular free wall cyst fistulized to the ventricular cavity 4)Ozer N, Aytemir K, Kuru G, et al. Hydatid cyst of the heart as a rare cause of embolization: report of 5 cases and review of publishedreports. J Am Soc Echocardiogr 2001;14:299-302. 5)De Martini M, Nador F, Binda A, Arpesani A, Odero A, Lotto A. Myocardial hydatid cyst ruptured in to the pericardium:cross-sectional echocardiographic study and surgical treatment. Eur Heart J 1988;9:819-824. 6)Pasaoglu I, Dogan R, Hazan E, Oram A, Bozer AY. Right ventricular hydatid cyst causing recurrent pulmonary emboli. Eur J Cardiothorac Surg. 1992;6:161-163. Gençpınar et al.