Asian Cardiovasc Thorac Ann 2002;10:259-261
© 2002 Asia Publishing EXchange Pte Ltd
Vascular Echinococcosis
Kamil Kaynak, MD,
Cengiz Köksal, MD1,
Kamuran Kazimo
lu, MD,
Cihan Ozbek, MD
Department of Thoracic and Cardiovascular Surgery Cerrahpasa Medical Faculty Istanbul University Istanbul, Turkey
1 Department of Cardiovascular Surgery Sureyyapasa Thoracic and Cardiovascular Disease Hospital Istanbul, Turkey
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For reprint information contact: Kamil Kaynak, MD Tel: 90 212 632 8474 Fax: 90 212 632 8474 email: kamil{at}istanbul.edu.tr Department of Thoracic and Cardiovascular Surgery, Cerrahpasa Medical Faculty, Istanbul University, Aksaray, Istanbul 34300, Turkey.
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ABSTRACT
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Three patients with arterial echinococcosis presented with chronic or acute arterial occlusion. One case diagnosed as Leriche’s syndrome was found at surgery to have aortic occlusion caused by hydatid cysts; arterial reconstruction was performed by aortoiliac interposition of a woven Dacron vascular graft. In the other 2 cases presenting with acute femoral or brachial artery occlusion, embolectomy revealed hydatid cysts as the cause of occlusion.
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INTRODUCTION
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Hydatid disease, caused by the tapeworm Echinococcus granulosus, is a parasitic condition with a worldwide distribution but found particularly in the Near and Middle Eastern Mediterranean countries of Europe and North Africa.1 Hydatid cysts may appear anywhere in the human body, but primary hepatic and lung localizations are the most frequent. Arterial localization of echinococcosis is quite rare and only a few cases have been reported previously.2,3
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CASE REPORTS
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CASE 1
A 50-year-old man was admitted to our clinic because of intermittent claudication, with a walking distance 50 to 100 meters, and impotence. He was a heavy smoker. Physical examination revealed an absence of femoral and distal pulses in both lower extremities. Contrast angiography showed total occlusion of the infrarenal aorta at the level of the bifurcation (Figure 1
). The right iliac artery at the level of the bifurcation and the left external iliac artery were visualized via collaterals from the iliolumbar and mesenteric arteries. A median laparotomy was executed to explore the infrarenal aorta. At the infrarenal level, the aorta was clamped proximally, the iliac arteries were clamped distally, and an aortotomy was performed. Multiple cysts were found inside the aorta, and removed completely (Figure 2). After resection of the involved aortic segment, the remaining aortic wall and periaortic area were sterilized with formalin to prevent recurrent hydatid cyst formation, and a 14 x 7-mm woven Dacron bifurcated interposition graft was placed. The postoperative course was uneventful, and distal pulses became palpable. Pathology of the cysts revealed hydatid disease. Whole-body computed tomography and echocardiography were undertaken to exclude hydatid disease in other locations. The patient was advised to take albendazole 10 mgákg-1áday-1 for 1 year. After 11 months of follow-up, he was asymptomatic.
CASE 2
A 27-year-old woman presented with bilateral lower extremity ischemia of abrupt onset soon after intra-abdominal surgery. Although it was 10 days since the symptoms of acute ischemia had started, both lower extremities were still viable with no necrosis or pares-thesia, suggesting good collateral circulation. Physical examination revealed cold and pale lower extremities with limited cyanosis of the toes of the right foot, and numbness in both legs. Femoral and distal pulses were absent. Contrast angiography showed distal aortic occlusion suggesting aortic saddle emboli (Figure 3). At surgery, bilateral femoral embolectomies were performed and thrombi and membranes were extracted from distal and proximal sites. Following revascularization, fasciotomy was carried out empirically for both legs, in view of the delayed embolectomy. Postoperatively, femoral, popliteal, and right distal pulses were palpable. Histopathology of the membranes revealed hydatid disease. Other disease localizations were excluded by computed tomography and echocardiography. The postoperative period was uneventful. On the 20th postoperative day, the patient was discharged on albendazole 10 mgákg-1áday-1 after closure of the fasciotomy incisions. At the 2-month follow-up, she was well and asymptomatic.
CASE 3
A 32-year-old woman from a suburban area of Istanbul was admitted because of acute onset of coldness and pain in the right arm. Physical examination was unremarkable except for the absence of right axillary, brachial, and distal pulses. Contrast angiography showed a distal right subclavian artery occlusion resembling an embolism. A right axillary embolectomy was performed, and thrombus and membranes were extracted. Postoperatively, the right axillary, brachial, radial, and ulnar pulses were palpable. Pathology of the membranes revealed hydatid disease (Figure 4). Whole-body computed tomography and echocardiography were unremarkable. The patient was discharged on albendazole 10 mgákg-1áday-1 for one year. Four months after the operation, she was well and free of symptoms.
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DISCUSSION
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Hydatid disease, or echinococcosis, is frequently encountered in sheep-rearing regions of the world, and remains a major economic problem in endemic areas. Once the infective eggs of the parasite are ingested, they are carried in the portal blood to the liver and after penetration, to the mucosa of the small intestine. They are mostly filtered out in the liver, with an incidence of hepatic involvement up to 80%.4 Quite a few reach the lungs, and this is the second most common site of lodgment of the parasite, with an incidence of 10% to 30%.1 However, some pass this second filtering system and reach the peripheral organs by the arterial blood stream.
In spite of the variability in location and clinical symptoms of hydatid disease, an arterial localization is rarely encountered. Oz and colleagues2 and Rada3 reported 2 cases of ischemia of the lower limb caused by hydatid disease. Although echinococcosis is rarely the causative factor in acute ischemia, embolectomy material should always be sent for histopathological examination in endemic areas, bearing in mind hydatid disease. Another form of arterial involvement of echinococcosis is intramural lodgment of the cysts, presenting as false aneurysm. There are a few reports of echinococcosis perceived as pseudoaneurysm of the thoracic aorta.5,6 Nevertheless, our case of distal aortic occlusion seems to be the first instance of hydatid disease mimicking Leriche’s syndrome reported so far.
Surgical intervention remains the definitive treatment for hydatid cysts in any location. Since primary vascular echinococcosis is seldom encountered, surgical therapy is individually tailored in such cases. Medical treatment with albendazole for 6 months after curative surgery is also highly recommended.6 Strict follow-up is required to detect any recurrence of the disease after surgical and medical treatment. These 3 cases emphasize that despite an absence of eosinophilia and other clinical manifestations of parasitic infection, in patients from sheep-rearing countries, primary vascular echinococcosis should be considered in the differential diagnosis of acute or chronic ischemia of the limbs.
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REFERENCES
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- Panagiotis NS, Aletras H. Hydatid disease of the lung. In: Shields RW, editor. General thoracic surgery. 4th ed. Philadelphia: Williams & Wilkins, 1994:1021–31.
- Oz N, Turkay C, Golbasi I, Dertsiz L, Sarper A, Demircan A, et al. Primary vascular echinococcosis: an uncommon cause of chronic iliofemoral arterial occlusion. Tex Heart Inst J
2000;27:209–11.[Medline]
- Rada IO. Ischaemic syndrome of the lower limb produced by a hydatid cyst. J Cardiovasc Surg
(Torino) 1986;27: 282–5.[Medline]
- Saidi F. Treatment of echinococcal cyst. In: Nyhus LM, Beker JR, Fsicher JE, editors. Mastery of surgery. 3rd ed. Philadelphia: Little, Brown & Company, 1998:1035–52.
- Zakhariev T, Stankev M, Baev B, Iliev R, Tschirkov A. A rare case of hydatid cyst perceived as false aneurysm of thoracic aorta. J Thorac Cardiovasc Surg
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- El Mesnaoui A, Bensaid Y, Ammar A, El Yagoubi M, Benabderrazik T, Benjelloun A, et al. False hydatid aneurysm of the thoraco-abdominal aorta [French]. J Chir
(Paris) 1996;133:222–5.
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ABSTRACT
INTRODUCTION
