Abstract
Cystic echinococcosis (CE) or hydatid disease is a zoonosis caused by ingestion of the eggs of the tapeworm Echinococcus granulosus. Larger cysts can cause symptoms by compressing surrounding tissues. Large cysts can also rupture and cause sudden onset of cough, fever, hypersensitivity reactions, and massive hemoptysis.
We report a case of hydatid cyst, which caused massive hemoptysis after an urgent percutaneous coronary intervention and was successfully controlled with bronchial artery embolization.
<Learning objective: In patients with hydatid cyst, even old lesions, there is a possibility of bleeding and massive hemoptysis after anticoagulation treatment. Bronchial artery embolization is a safe and effective therapy in these situations, when the patient is not a good surgical candidate and the bleeding is life-threatening.>
Keywords: Embolization, Hemoptysis, Hydatid cyst, Acute coronary syndrome
Introduction
Cystic echinococcosis (CE) or hydatid disease is a zoonosis caused by ingestion of the eggs of the tapeworm Echinococcus granulosus. E.granulosus tapeworms live in the intestine of carnivores such as dogs and wolves (definite host). The infected carnivores pass out the eggs by defecation. The eggs are ingested by an intermediate host (herbivores such as goats, pigs, cattle, or sheep). Humans can also act as intermediate hosts if they ingest the eggs. After ingestion, eggs hatch into embryos in the intestine, penetrate the intestinal mucosa, spread through portal venous system and lodge in different tissues, mainly liver and lungs as the main filtering organs. Then embryos transform to hydatid cysts [1,2]. Small pulmonary cysts may remain asymptomatic for a long time. But larger cysts can cause symptoms by compressing surrounding tissues. Large cysts can also rupture and cause sudden onset of cough, fever, hypersensitivity reactions, and massive hemoptysis [3–5].
We report a case of hydatid cyst, which caused massive hemoptysis after an urgent percutaneous coronary intervention (PCI) and was successfully controlled with bronchial artery embolization (BAE).
Case report
A 42-year-old chronic smoker and intravenous drug abuser presented to the emergency department with typical retrosternal chest pain, radiating to the left arm. He reported a history of hepato-pulmonary hydatid cysts, for which he had thoracic surgery and he was also taking albendazole and praziquantel since then (20 months). He had positive family history of ischemic heart disease. His blood pressure was 145/90 mm/Hg and heart rate was 110 per minute. His physical examination was unremarkable except for crackles on the left lower lobe field and a thoracotomy scar on the chest. In the initial electrocardiography (ECG), the patient had sinus tachycardia, and there were flat ST-segment depressions (2 mm) in precordial leads. He was admitted to the coronary care unit (CCU) with the diagnosis of acute coronary syndrome (ACS). He was prescribed full standard medical therapy for ACS (including aspirin, heparin, clopidogrel etc.). Laboratory studies revealed an elevated cardiac troponin I and creatine kinase-MB. Platelet count, prothrombin time, partial thromboplastin time, and international normalized ratio were within normal range. At bedside echocardiography in CCU, left ventricular (LV) size was normal, LV ejection fraction was 45%, regional wall motion abnormality (RWMA) was observed in anterolateral wall of LV, and peak pulmonary artery pressure was 33 mmHg. Size and function of right ventricle were normal. He underwent urgent coronary artery catheterization. In catheterization, there was an 80% stenosis in the proximal part of left anterior descending artery (LAD) with moderate run off. PCI was performed on LAD using Xience V 3.5 mm × 23 mm stent (Abbott Vascular, Santa Clara, CA, USA). Because the patients was at high risk of stent thrombosis and a clot was observed in distal left main coronary artery during catheterization, he was administered eptifibatide and intravenous heparin (in addition to standard full doses of aspirin and clopidogrel) after the procedure. After two days, he experienced massive hemoptysis. After initial resuscitation, we reviewed his past medical history again. Evaluation of his medical record revealed that on that time his spiral chest computed tomography (CT) scan had shown a left lower lobe cavitary lesion with septae, air-fluid level and ruptured internal membrane seen as floating structures, in favor of complicated and ruptured hydatid cyst (Fig. 1). Then he had undergone thoracotomy, and wedge resection of the left lower lobe and obligating terminal bronchioles ending in the cyst bed had been performed. He had been discharged with albendazole and praziquantel. The patient had intermittent sputum with blood streaks (non-massive hemoptysis). His last chest CT scan after 18 months had shown ill-defined mass in left lower lobe associated with cystic lesion in right liver lobe (Fig. 2). Although the pulmonary lesion had not been calcified and the infectious disease specialist had recommended a second surgical intervention, the patient had not given consent for surgery and chose to continue medical therapy.
Fig. 1.
Chest computed tomography scan of the patient at initial presentation of hydatid disease.
Fig. 2.
Last follow-up chest computed tomography scan of the patient, obtained 2 months prior to presentation with massive hemoptysis.
In his index massive hemoptysis after PCI, a pulmonologist and a thoracic surgeon were consulted urgently. After comprehensive assessment of the patient's status and risk profile for any surgical intervention, they suggested that emergency angioembolization of the bleeding artery should be done. The patient underwent selective and nonselective bronchial angiography from right femoral artery access. Two bronchial arteries (with tortuosity, enlargement, and abnormal blood flow) originating from intercostal arteries (distal to the location of the previous hydatid cyst) were determined as the source of bleeding (Fig. 3) and they were embolized successfully with polyvinyl alcohol (PVA) particles (diameter of 500–710 µm). The hemoptysis was stopped completely after 6 h and he was discharged uneventfully after 5 days. During the follow-up visits after 3 months, he had not experienced any hemoptysis, despite double antiplatelet therapy (aspirin and clopidogrel).
Fig. 3.
Bronchial artery angiogram, demonstrating bronchial artery enlargement, tortuosity, and abnormal blood flow.
Discussion
Surgery is the mainstay of therapy of hydatid cysts in patients who are able to undergo surgery. Recurrence is usually due to inadequate cyst removal, spillage of cyst contents intraoperatively, or undetected cysts during surgery [2,4]. Hydatid cysts can rarely present with massive hemoptysis. Massive hemoptysis, defined as expectoration of greater than 200–600 ml of blood per day, [6] is a devastating and life-threatening condition, requiring timely intervention. Conservative management of massive hemoptysis has an estimated mortality rate of 50 to 85% [7]. The reported mortality rates for surgery performed for massive hemoptysis is 40%, when the surgery is undertaken as an emergency procedure [8]. We believe that our patient would have an even higher mortality risk owing to his recent myocardial infarction, PCI, and anticoagulant therapy. BAE effectively controlled the bleeding and stabilized his condition.
BAE is an acceptable procedure in the management of massive hemoptysis and immediate success rate for cessation of bleeding is high (85%) [8,9]. Even in patients who are candidates for surgical management of hemoptysis, BAE is effective for preparing the patient for elective rather than emergency surgery [10].
Conclusion
In patients with hydatid cyst, even old lesions, there is a possibility of bleeding and massive hemoptysis after anticoagulation treatment. BAE is a safe and effective therapy in these situations, when the patient is not a good surgical candidate and the bleeding is life-threatening. It acts as a palliative therapy, allowing the physicians to treat the underlying condition by appropriate surgical or medical therapy thereafter.
Declaration of Competing Interest
The authors declare that there is no conflict of interest.
Funding source
None.
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