ruptured cardiac hydatid cyst

Sonderdruck aus / reprint from

VASA

Zeitschria fiir Gefasskrankheiten Journal for vascular Direases

Verlag Hans Huber Bern Gdttingen Seattle Toronto

H.Ceyran et al. A rare cause of peripheral arterial embolism

VASA 2002; 31:129-131

Department of Thoracic and Cardiovascular Surgery1and Department of Radiodiagnostics2, Erciyes University, Kayseri, Turkey

A rare cause of peripheral arterial embolism: ruptured cardiac hydatid cyst H. Ceyranl, K. Tasdemirl, T Tezcanerl, E Asgun1, 0 . I. Karahanz, 0.N. Emirogullanl and H. Andacl

Summary

Zusammenfassung

One of the important unfavorable events that occurduring the course of the cardiac hydatid cyst is rupture of the cyst and embolism of the germinative membrane. Peripheral arterial embolism of this germinative membrane is uncommon but is a potential risk due to the nature of the disease. Ruptured cardiac hydatid cyst should be suspected in young patients who have a peripheral arterial embolism and come from sheep-raising areas andor if they have a suspected embolectomy material resembling germinative membrane. Following the embolectomy and reconstruction of the circulation in the involved extremity, ruptured cardiac hydatid cyst should be diagnosed immediately and excision of the cardiac cyst should be pe$ormed as quickly as possible. In this case report, we present two patients who had lower extremity embolism originating from the ruptured cardiac hydatid cyst and were operated on for cardiac cyst excision.

Eine seltene Ursache einer arteriellen Embolie: Ruptur einer kardialen Echinokokkenzyste Eine der besonders komplikationstrachtigenBegleiterscheinungen der kardialen Echinokokkose ist die Ruptur der Zyste und Embolisierung der Keimmembran. An eine rupturierte Echinokokkenzyste sollte bei jungen Patienten mit arterieller Embolie gedacht werden, die aus einer Gegend stammen, in der Schafzucht betrieben wird. Das embolektierte Material ahnelt einer Keimmembran. Die Sanierung der kardialen Emboliequelle mit Exzision der Zyste sollte schnellstmoglich nach Wiederherstellungder peripheren Zirkulation erfolgen. Wir berichten hier uber den Verlauf von zwei Patienten mit diesem seltenen Krankheitsbild.

Introduction

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Peripheral arterial embolism originating from the heart is not uncommon. The heart is the source of embolism in 90% to 96% of the cases published in the literature [4]. Most observed embolism material is thrombus frequently caused by severe cardiopathy including chronic atrial fibrillation, valvular pathology and ischemic heart disease. Not only thrombus but also other rare circumstances originating from the heart may cause peripheral arterial embolism. Ruptured cardiac hydatid cyst is one of these rare causes. Cardiac hydatid cysts have an incidence ranging from 0,02% to 2% [l 11 may be ruptured, resulting in peripheral embolism of germinative membrane and spillage of cyst's contents. The germinative membrane may locate anywhere throughout the arteries. Many different clinical features occur in respect of the occluded artery. These vary from stroke [I], severe pulmonary embolism [3] to minor limb embolism and may cause death or majorlminor sequelas. Peripheral arterial embolism originating from the ruptured cardiac hydatid cyst is uncommon and causes only limb ischemia that can be easily treated by minor surgery but has a potential risk due to the nature of the Submitted 8. 10. 2001 1Accepted after revision 21. 10. 2001

Key words Arterial embolism, cardiac hydatid cyst

disease. It should be considered as a sign of underlying severe heart disease. In this case report, we present two patients who had lower extremity embolism originating from the ruptured cardiac hydatid cyst and were operated on for cardiac cyst excision.

Case report 1 A 27 year-old male was admitted to the emergency services of Erciyes University Medical Faculty in Turkey. He has been having severe pain on the right lower extremity for about six hours. Physical examination revealed an ischemic color on skin below the knee level and this portion of the leg was cold. Right dorsalis pedis and tibialis posterior pulses were absent while right popliteal pulse was palpated. When the patient's clinic records were reviewed it was learned that he had two operations 6 and 4 year ago for right temporal intracerebral hydatid cyst. The patient underwent an emergency operation for peripheral arterial embolism caused by the rupture of a suspected cardiac hydatid cyst. Embolectomywas performed to the superficialfemoral artery by a 3 F Fogarty balloon catheter. The material con-

129

H. Ceyran et al. A rare cause of peripheral arterial embolism

sisting of white and thin membranous tissue and fresh clot was removed (Fig. 1). All distal pulses were palpable in the course of an uncomplicated postoperative period. Pathologic examination of the removed material revealed that it was germinative membrane of a hydatid cyst and echocardiographic examination exposed a septated cyst located in the posterior wall of the left ventricle around the posterior papillary muscle 2 x 2 cm diameter and another inferolaterallybetween the pericardial layers 7x7 cm diameter. Ruptured cardiac hydatid cyst was diagnosed by these laboratory findings and the patient's clinic history. Then it was decided that cardiac surgery for cyst excision was required. The patient was operated on 25 days after embolectomy. Operation was carried out through a sternotomy. The pericardium was opened anteriorly and cardiopulmonary bypass instituted after the ascending aorta and bicaval canulation was achieved. The heart was arrested by cold crystalloid cardioplegic solution infusion through the aortic root and topical cooling by ice slush following the aortic cross clamping. The cyst was located on the posterior wall of the left ventricle (Fig. 2) extending laterally into the pericardium and was exposed. The cystic content was aspirated with a needle connected to a suction pump and the cavity was irrigated with hypertonic saline solution. Hydatid membrane was removed carefully to avoid any spillage of the content. The germinative membrane of the hydatid cyst was enucleated and the pericystic layer was excised leaving a 0,5 cm fibrous rim. The resultant defect on the posterior wall was closed by horizontal mattress sutures reinforced by Teflon pledgets. The patient had to have a re-exploration due to excessive mediastinal bleeding in the postoperative period. Except for this unpleasant complication, we did not experience any unfavorable event in the postoperative course. Treatment with mebendazole was recommended to the patient for two months postoperatively and he was discharged 17 days after surgery. He has been well up to now.

VASA 2002; 31: 129-131

Fig. 1: Germinative membran which was remowed from superficial femoral artery.

Fig. 2: Intraoperative view of ruptured hydatid cyst.

Case report 2

130

A 24-year-old male patient was admitted to the emergency services with severe pain in his lower extremities for about five hours. On clinical examination, his lower extremities were found to be cold and femoral pulses were absent bilaterally. The patient was operated on immediately and bilateral femoral embolectomy was carried out in the same manner explained for the previous patient. The material removed from iliac arteries resembled germinative membrane. After the procedure, echocardiogramwas performed which revealed an intramyocardial cyst of 3 x 3 cm diameter (Fig. 3), lying through the intraventricular septum of the left ventricle. The patient underwent a cardiac operation with the diagnosis of ruptured cardiac hydatid cyst at the same day. Open heart surgery was performed and a, ruptured and thrombosed hydatid cyst was seen on the intraventricular septum after the left ventriculotomy. The cystic content and fresh thrombus was aspirated and the cavity was irrigated. The hydatid membrane was removed. he germinative

Fig. 3: Echocardiographic view of ruptured hydatid cyst.

H. Ceyran et al. A rare cause of peripheral arterial embolism

membrane was enucleated and the pericystic layer was excised. The resultant defect on the interventricular septum was closed by horizontal mattress sutures reinforced by Teflon pledgets. The left ventriculotomy was closed and the patient was removed from the cardiopulmonarybypass. Histopathologic diagnosis confirmed a hydatid cyst. The postoperative period was uncomplicated. Treatment with mebendazole was recommended to the patient for two months. He was discharged 7 days after surgery. This patient has also been well up to now.

Discussion Hydatid cyst of the heart is uncommon, its incidence ranging from 0,02% to 2% in literature [7, l I]. The liver is the most frequently involved location (more than 65%) while the lungs are involved in 25% of cases [7]. Cardiac hydatid cysts can be found in any localization of the heart. Hydatid cyst is a parasitic infection caused by Echinococcus granulosus. Definitive hosts of this cestod are dogs and other canines while ungulates (both domestic and wild) usually sheep are intermediate hosts. The place of the human in the life cycle of Echinococcus granulosus is as an incidental intermediate host [7]. The infectious agent usually reaches into the myocardium via the coronary circulation and the cyst is formed in 1 to 5 years [7]. Through the course of disease, myocardial reaction occurs and a fibrous adventitial pericystic layer surrounds the laminated membrane of the cyst named the germinative membrane. The disease is usually seen in the people from sheep-raising areas. Manifestations and clinical features including chest pain, congestive heart failure, embolism [7], valvular syndrome [5, 91, ischemic syndrome [13], arrhythmias, conduction disturbances and sudden cardiac death have been observed in these patients. One of the important unfavorable events that may occur during the course of cardiac hydatid cyst is rupture of the cyst and embolism of the germinative membrane. Spillage of the cyst's content may cause disseminated cystic disease and also anaphylactic shock [6]. Any level of the arterial tree can be occluded by germinative membrane from the hydatid cyst. The level involved by germinative membrane is important because death or serious sequelae may occur if it occludes one of the arteries perfusing the brain or other important organs [I-3,8, 10, 121while only an extremity ischemia may occur if it settles into a limb artery. Peripheral arterial embolism originating from the ruptured cardiac hydatid cyst causes upper or lower limb ischemia in respect of the level of the occluded artery. If it involves the terminal aorta, bilateral lower limb ischemia occurs [lo] and requires bilateral femoral embolectomy. If its localization is more distal, ipsilateral embolectomy should be performed. At operation, care should be focused on the material removed from the artery especiallyin young patients from sheep-raising areas. If there is suspected material resembling germinative membrane, echocardiography should be performed immediately. Echocardiography generally exposes the cyst(s), in any diameter and in

VASA 2002; 31:129-131

any localization inside or around of the heart. Non-specific alterations may be seen in the ECG. The skin test of Casoni may be helpful. Serologic tests like latex agglutination test and immunoelectrophoresis may be used to diagnose a hydatid cyst occurrence [7]. Rapid histopathologic evolution of the material removed from the artery is a valuable diagnostic criterion to detect the presence of the ruptured cardiac hydatid cyst. Cardiac surgery for cyst excision should be planned and performed as quickly as it can be done. Mebendazole treatment to eradicate the remnant cestods should be recommended to all patients for aperiod postoperatively. These patients should be closely followed-up against the recurrence of the disease. In conclusion, ruptured cardiac hydatid cyst should be suspected in young patients who have a peripheral arterial embolism and come from sheep-raising areas andlor if they have suspected embolectomy material resembling germinative membrane. Echocardiography and rapid histopathologic evolution of the material removed from artery should be done immediately and if cardiac hydatid cyst is confirmed, excision of cardiac cyst should be performed in early period. Early intervention in these patients can be life saving.

References 1 Benomar A, Yahyaoui M, Birouk N et al. Middle cerebral artery occlusion due to hydatid cysts of myocardial and intraventricular cavity cardiac origin. Two cases. Stroke 1994; 25: 886-8. 2 Byard RW, Bourne AJ. Cardiac echinococcosis with fatal intracerebral embolism. Arch Dis Child 1991; 66: 155-6. 3 Ege E, Soysal 0 , Gulculer M et al. Cardiac hydatid cyst causing massive pulmonary embolism. Thorac Cardiovasc Surg 1997; 45: 249-50. 4 Haimovici H. Arterial embolism of the extremities and technique of embolectomy. In: Haimovici H, Ascer E, Hollier LH, Strandness DE, Towne JB. Haimovici's Vascular Surgery, Fourth Edition, Massachusetts; Blackwell Science, 1996: 423-44. 5 Hazan E, Leblanc J, Robillard M et al. Hydatid cyst of the right ventricle revealed by an acute complication: emergency exeresis with prosthetic replacement of the tricuspid valve. Chirurgie 1970; 96: 257-60. 6 Madariaga I, de la Fuente A, Lezaun R et al. Cardiac echinococcosis and systemic embolism. Report of a case. Thorac Cardiovasc Surg 1984; 32: 57-9. 7 Miralles A, Bracamonte L, Pavie A et al. Cardiac echinococcosis. Surgical treatment and results. J Thorac Cardiovasc Surg 1994; 107: 184-90. 8 Oguzkaya F, AkcaliY, Kahraman C, Emirogullan 0 N, BilginM. Unusually located hydatid cysts: Intrathoracic but extrapulmonary. Ann. Thorac Surg 1997; 64: 334-7. 9 Perez-Gomez F, Duran H, Tamames S et al. Cardiac echinococcosis: clinical picture and complications. Br Heart J 1973; 35: 1326-31. 10 Rosenberg T, Panayiotopoulos YP, Bastounis E et al. Acute abdominal aorta embolism caused by primary cardiac echinococcus cyst. Eur J Vasc Surg 1993; 7: 582-5. 11 Rossouw GJ, Knott-Craig CJ, Erasmus PE. Cardiac echinococcosis: cyst removal in a beating heart. Ann Thorac Surg 1992; 53: 328-9. 12 Turgut M, Benli K, Eryilmaz M. Secondary multiple intracranial hydatid cysts caused by intracerebral embolism of cardiac echinococcosis: an exceptional case of hydatidosis. Case report. J Neurosurg 1997; 86: 714-8. 13 Vestri A, Nigri A, Massi L et al. Electrocardiographic picture of myocardial infarct during echinococcosis of the heart. Boll Soc Ital Cardiol 1972; 17: 7 5 2 4 .

Hakan Ceyran, MD, Erciyes University Medical Faculty, Department oj'Thoracic and Cardiovascular Surgery, 38030 ~ a ~ s e rTurkey i, E-Mail: [email protected]

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Vasa is the European journal of vascular medicine. It is the official organ of the German, Swiss, Czech, and Slovenian Societies of Angiology. The journal publishes original research articles, case reports and reviews on vascular biology, epidemiology, prevention, diagnosis, medical treatment and interventions for diseases of the arterial circulation, in the field of phlebology and lymphology including the microcirculation, except the cardiac circulation. Vasa combines basic science with clinical medicine making it relevant to all physicians interested in the whole vascular field. Download the preliminary program of the 16th tri-country congress Angiology 2013 in Graz (PDF in German). Information for authors Submit your manuscript here. Instructions to authors (PDF)

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