CASE REPORTS One-Stage Surgical Treatment of Cardiac and Pulmonary Echinococcosis Alejandro Aris, M.D., Carlos Leon, M.D., Jose 0. Bonnin, M.D., Constantino Serra, M.D., and Jose M. Caralps, M.D. Q waves and negative T waves in leads I and aVL, suggestive of myocardial necrosis of the high lateral wall (Fig 2). Laboratory tests were essentially normal except for mild eosinophilia and a positive serological test for hydatid cyst (latex agglutination test). The echocardiogram indicated the presence of a mass near the base of the aorta. The patient underwent cardiac catheterizaCardiac echinococcosis represents an infre- tion. Left ventricular end-diastolic pressure was quent complication of hydatid disease but its 18 mm Hg. Left ventriculogram showed hyeffects can be devastating if not treated aggres- pokinesia of the lateral wall. Coronary artesively. The purpose of this article is to report riogram revealed no obstructing lesions but the case of a young woman with previously the two branches of the left coronary artery treated hepatic echinococcosis in whom pul- were displaced by a mass, which created a monary and cardiac hydatid cysts developed. "cold zone" between them (Fig 3). The patient underwent successful surgical reThe diagnosis of left ventricular hydatid cyst moval of the cysts in a one-stage operation with and left lower lobe hydatid cysts was made, and cardiopulmonary bypass. the patient underwent operation. The chest was entered through a transsternal bilateral thoraA 26-year-old woman was admitted to another cotomy, which provided ample exposure of the hospital because of shortness of breath and car- heart and both pleural cavities. While the aorta diac palpitations following mild exercise. The and venae cavae were being cannulated for carepisode was diagnosed as supraventricular diopulmonary bypass, a rapid supraventricular tachycardia and was treated by electric counter- tachycardia developed. Once the patient was on shock, which was successful. Pertinent past the pump, the pericardium was freed from the history included two abdominal operations be- lateral, upper aspect of the left ventricle where cause of hepatic hydatid cysts, 15 and 7 months an egg-sized mass, covered by some myocardial previously. A routine chest roentgenogram was fibers, was bulging. The cyst was punctured, and its contents found to be abnormal. The diagnosis of cardiac and pulmonary echinococcosis was suggested, were aspirated with a syringe. A creamy yellow and the patient was transferred to our hospital material was obtained. Then the operative field for further workup and therapy. Chest roent- was covered with gauze pads moistened with genogram made at admission (Fig 1)revealed a 3% saline solution. The cyst was opened, and mass at the left heart border as well as a solitary a large number of ruptured membranes of bilocular nodule at the posterior aspect of the daughter cysts were removed. The cavity was left lower lobe. The electrocardiogram showed emptied, and most of the fibrous pericyst was excised. The deepest part was left untouched From the Cardiac and Thoracic Surgery Units, Hospital de since there was only a thin layer of myocardium la Santa Cruz y San Pablo, Barcelona, Spain. between it and the left ventricular cavity. The Accepted for publication June 10, 1980. heart was defibrillated, and while the patient Address reprint requests to Dr. Aris, Chief, Cardiac Sur- was still on the pump, the entire left lung was gery Unit, Hospital de la Santa Cruz y San Pablo, Avda S. retracted medially. A bilocular mass was idenAntonio M. Claret 167, Barcelona 25, Spain. ABSTRACT The case of a 26-year-old woman with cardiac and pulmonary echinococcosis is presented. Surgical treatment of the intrathoracic disease was done in a one-stage operation. Under cardiopulmonary bypass, a left ventricular hydatid cyst and a lower lobe pulmonary cyst were removed. The patient is completely asymptomatic and is leading a normal life two years after operation. 564 OOO3-4975/81/060564-05$01.25 @ 1980 by The Society of Thoracic Surgeons 565 Case Report: Aris et al: Cardiac and Pulmonary Echinococcosis A B Fig 1. (A) Posteroanterior chest roentgenogram shows a mass at the left heart border and a nodule at the left lung field. (B)Lateral v i e w shows this nodule located posteriorly (arrow). tified in the lower lobe and excised en bloc. Following the cystectomy, capitonnage of the residual cavity with interrupted sutures of absorbable material was performed. Cardiopulmonary bypass was discontinued and after careful hemostasis, the chest was closed. The patient made an uneventful recovery and was discharged on the tenth postoperative day. Follow-up at regular intervals reveals that she has not had any further arrhythmias and is leading a normal, active life. Pathological examination of both specimens Fig 2 . Electrocardiogram shows Q waves and negative T w a v e s in leads 1 and aVL, which suggest necrosis of the high lateral wall. I II Ill aVR aV L aVF v1 v2 v3 v4 VS V6 566 The Annals of Thoracic Surgery Vol 31 No 6 June 1981 Fig 3 . Left c o r o n a y arteriogram in left anterior oblique projection. The left anterior descending corona y a r t e y is displaced upward and the circumflex corona y arte y , posteriorly. There is an avascular zone in between them. was consistent with hydatid cyst. Necrotic material was found in both cavities but no scolices were seen. occurred in our patient, in whom hepatic, pulmonary, and cardiac hydatid cysts developed in the short interval of 15 months. The fate of a cardiac echinococcal cyst is usually rupture, either in the pericardial cavity [21, 221 or in a cardiac chamber (38% of the cases according to Di Bello and Menendez [7]). When the latter occurs, death of the patient by anaphylactic shock or massive embolization to different organs [ll, 22-24] can ensue. Cardiac hydatid cysts rupture more frequently than do hydatid cysts in other organs, probably because of the constant motion. Also, rupture in the right ventricle is more frequent than in the left (88% versus 33%) [71 because of the thicker wall and higher pressure in the latter. Other events in the life of a cyst include rupture inside the adventitia with formation of daughter cysts, and interference with the conduction system of the heart, thereby inducing arrhythmias [25]. Both were found in our patient. Diagnosis of cardiac hydatid disease is difficult, especially in the early stages. Roentgenographic findings can be consistent with an aneurysm 15, 261. Although several changes in the electrocardiogram are said to be characteristic of cardiac echinococcosis [2, 8, 141 and helpful in distinguishing it from ventricular aneurysm, our patient showed Q waves in some leads, indicating myocardial necrosis, an unusual feature in cardiac hydatid disease. Echocardiography has proved a valuable diagnostic test in detecting the location of possible cardiac cysts [18], as it did in our patient. Cardiac catheterization with cineangiography is the most reliable test for an accurate diagnosis. Coronary angiography also should be performed since coronary compression by a growing cyst has been reported [17].In our patient, the lumen of the arteries was patent but the cyst had displaced the two branches of the left coronary artery, thereby creating an avascular zone between them. This angiographic finding was described in 1976 [271 and 1979 Comment Different reports [l-31 estimate that 0.5 to 2% of all cases of hydatid cysts involve the heart. Most of the cases occur in countries where the disease is endemic-Uruguay [l,4-81, Argentina 191, Iran [lo, 111, Greece [12,131, Israel [14,151, and Spain. In the last named, more than 20 cases have been reported, most of them by surgical teams [16-201. Implantation of the hexacanth embryo in the myocardium usually occurs after the embryo has passed the pulmonary capillary network and has reached the heart by way of the coronary circulation. This explains the higher incidence of echinococcal cysts in the left ventricle and ventricular septum, which have the richest blood supply. Before it reaches the heart, the embryo of the Echinococcus grunulosus has filtered through the sinusoids of the liver (the 1201. Cardiac echinococcosis should be treated most commonly involved organ) and the pulmonary circulation. The embryo may be im- surgically as soon as the diagnosis is estabplanted in these organs, developing an hydatid lished because of the fatal complications that (or echinoccocal) cyst. This sequence of events can occur. Intervention involves the removal of 567 Case Report: Aris et al: Cardiac and Pulmonary Echinococcosis the cyst (or cysts), including the adventitia. Our patient has recovered completely and is Needle aspiration prior to the surgical excision free from hepatic, pulmonary, and cardiac is advisable and if clear liquid is obtained, the echinococcosis two years following the incyst should be injected with a sterilizing solu- trathoracic procedure. tion (hypertonic saline, ether, formaldehyde) before it is opened. Although most of the reported patients with References 1. Dighiero J, Canabal EJ, Aguirre CV, et al: cardiac echinococcosis were operated on Echinococcus disease of the heart. Circulation through a.left thoracotomy without the aid of 17:127, 1958 cardiopulmonary bypass [3, 10, 12-15], the 2. Murphy TE,Kean BH, Venturini A, Lillehei CW: low-risk perfusion technology now available Echinococcus cyst of the left ventricle: report of a case with review of the pertinent literature. J makes the use of extracorporeal circulation adThorac Cardiovasc Surg 61:443,1971 visable. 3 . Ramos G, del Villar JL, Sainz JL, et al: Regarding pulmonary hydatid cysts, the Hidatidosis cardiaca. Rev Clin Esp 121:411,1971 history of our patient is fairly representative. 4. Artucio H, Roglia JL, Di Bello R, et al: Hydatid The disease usually is located in one lobe. In a cyst of the interventricular septum of the heart series of 100 patients reported by Aytac [28], the ruptured into the right ventricle. J Thorac Cardiovasc Surg 44:110,1962 distribution of single-lobe involvement was 5. Di Bello R, Rubio R, Dighiero J, et al: Pseudoabout the same for the left upper and both aneurysmatic form of cardiac echinococcosis: relower lobes. Symptom-free patients are rare port of a new case and review of the literature. J (about 5%) [28, 291, and pulmonary involveThorac Cardiovasc Surg 45:657,1963 ment in our patient prabably would have 6. Di Bello R, Ab6 JC, Dubra J, Diaz EG: Hydatid cyst of the heart. J Thorac Cardiovasc Surg passed undetected if the cardiac cyst had not 46:522,1963 developed. Enucleation of the cyst [30] followed 7. Di Bello R, Menendez H: Intracardiac rupture of by capitonnage of the cavity is the preferred hydatid cysts of the heart: a study based on three treatment for small, noncomplicated Echinococpersonal observations and 101 cases of the world cus cysts of the lung [28]. literature. Circulation 27:366, 1963 The case of our patient deserves some par8. Canabal EJ, Dighiero J, Purcallas J, et al: Echinococcus disease of the left ventricle: a cliniticular comments. The first symptoms of the cal, radiological and electrocardiographic study. disease were arrhythmias, a rare presentation. Circulation 12:520,1955 According to Heyat and associates 1101, who re9. Gonzalez Bosch R, Mosto D: Hidatidosis carviewed the signs and symptoms in 82 patients diaca. Prensa Med Argent 24:38,1937 with cardiac hydatid cysts, only 11%had “pal- 10. Heyat J, Mokhtari H, Hajaliloo J, Shakibi JG: Surgical treatment of echinococcal cyst of the pitation of the heart.” heart. J Thorac Cardiovasc Surg 61:755,1971 The existence of pulmonary and cardiac echinococcosis in the same patient has been re- 11. Handjani AM, Farpour A, Mechanic K, et al: Cardiovascular echinococcosis. Am J Surg ported previously [ll, 181 but, we believe that 117:666,1969 we are among the first to remove both cysts in a 12. Karageorgis B, Papanicolis I: Some remarks on one-stage operation. A second operation to retwo personal cases of cardiac echinococcosis. Dis Chest 51:199,1967 move the lung cyst was avoided. Although there is a recent trend to use me- 13. Papamichael E, Ikkos D, Milingos M, Yannacopoulos J: Echinococcosis of the heart. Chest dian sternotomy for pulmonary operation, this 59:280,1971 approach has been questioned for lesions in- 14. Romanoff H, Milwidsky H: Primary echinococvolving the left lower lobe [31]. For this reason, cosis of the heart cured by operation. J Thorac Cardiovasc Surg 43:677, 1962 we decided to use a transverse transsternal bilateral thoracotomy, which provided excellent ex- 15. Romanoff H: Echinococcosis of the heart: report of three new cases. J Thorac Cardiovasc Surg posure of the heart as well as of the posterior 66:29,1973 aspect of the left lower lobe. Under cardiopul- 16. Calvo Melendro J:Quistes hidatidicos de coramonary bypass, the entire left lung was colz6n. Rev Clin ESP 42:397,1951 lapsed and the pulmonary cyst easily excised. 17. Rivera R, Delcan JL: Surgical treatment of coro- 568 The Annals of Thoracic Surgery Vol 31 No 6 June 1981 nary insufficiency produced by cardiac echinococcosis. Chest 78:849, 1980 18. Soler-Soler J, Noguera L, Fit0 R: Recent multiple pulmonary nodules with progressive cardiomegaly. Chest 73:397, 1978 19. Tellez G, Nojek C, Juffe A, et al: Cardiac echinococcosis: report of 3 cases and review of the literature. Ann Thorac Surg 21:425, 1976 20. Montoyo JV, Nogues-Antich FJ, Rivas L, et al: Left ventricular echinococcosis diagnosed by coronary cineangiography. Eur J Cardiol 10:215, 1979 21. Di Bello R, Stanham J, Rubio R, Muxi F: Hydatid cyst of the left ventricular wall with rupture into the intrapericardial space. J Thorac Cardiovasc Surg 44:268, 1962 22. Purriel P, Perez Fontana V, Muras 0, Mendoza D: Equinococosis cardiaca con siembra pericardica y de pequeiio y gran circulo. Torax 2:188, 1953 23. Dumolard MM, Lemaire C: Kyste hydatidique de l’oreillette droite. Alger Med 32:293, 1928 24. Purriel P, Muras 0, Tomaluro D, Mendoza D: 25. 26. 27. 28. 29. 30. 31. Equinococosis cardiaca: siembra metastatica pulmonar. Torax 1:223, 1952 Peters JH, Dexter L, Weiss S: Clinical and theoretical considerations of involvement of the heart with Echinococcus cysts. Am Heart J 29:143, 1945 Hatzeganu I, Vasiliu T, Moga A: Le kyste hidatidique du coeur simulant un anevrisme. Arch Ma1 Coeur 29:72, 1936 Fawzy ME: Hydatid disease of the heart. Br Heart J 38:307, 1976 Aytac A, Yurdakul Y, Ikizler C, et al: Pulmonary hydatid disease: report of 100 patients. Ann Thorac Surg 23:145, 1977 Sarsam A: Surgery of pulmonary hydatid cysts: review of 155 cases. J Thorac Cardiovasc Surg 62:663, 1971 Barret NR: Removal of simple univesicular pulmonary hydatid cyst. Lancet 2:234, 1949 Urschel HC Jr: Discussion of Cooper JD, Nelems JM, Pearson FG: Extended indications for median sternotomy in patients requiring pulmonary resection. Ann Thorac Surg 26:413, 1978
