J Thromb Thrombolysis DOI 10.1007/s11239-014-1147-5

Rare reason for pulmonary embolism: one case of pulmonary hydatid cyst and review of the literature Zhao Yuan • Muhebaiti Maihesuti • Liu Wenya • Zhou Cheng • Nuer Biyemu Wang Haitao



Ó Springer Science+Business Media New York 2014

Abstract Pulmonary embolism due to hydatid disease has been rare clinically. Here we present a patient case who suffered from multiple cystic hydatidosis within bilateral lung parenchyma and complicated bilateral pulmonary embolisms. The case was a 70-year-old man who complained of an increasingly severe cough and hemoptysis post-operation of hepatic hydatid cyst. Chest radiographs showed that the patient had multiple nodules in the bilateral chest. Computed tomography (CT) depicted that some lesions were multivesicular cysts and some consisting of sophisticated complications. CT pulmonary angiography revealed bilateral pulmonary arterial embolisms. T2weighted magnetic resonance images (MRI) clearly demonstrated the daughter cysts inside the lesions with high intensive signal. Echinococcosis serologic testing was positive. Thus, the pulmonary embolism was caused by hydatid cyst based on the imaging findings and serologic test. Keywords Hydatid cyst  Pulmonary embolism  Complication  Imaging modalities

Z. Yuan  M. Maihesuti (&)  L. Wenya (&)  Z. Cheng  N. Biyemu  W. Haitao Imaging Center, First Affiliated Hospital, Xinjiang Medical ¨ ru¨mqi 830054, Xinjiang, University, 137 Liyushan Road, U China e-mail: [email protected] L. Wenya e-mail: [email protected] Z. Yuan e-mail: [email protected]

Introduction Echinococcosis is caused by the larval forms of echinococcus granulosus, which requires two mammalian hosts to complete its life cycle [1]. The definitive hosts are primarily canines in which the adult tapeworms inhabit the small intestines and excrete tapeworm eggs that contaminate food, vegetables, and/or water. Humans serve as accidental hosts suffering the echinococcosis through ingestion of contaminated substances. In humans, cystic echinococcosis (CE) is one of the most important chronic helminthic diseases that have an initial asymptomatic incubation period of 5–15 years followed by a chronic progress of symptomatic course. The process and outcomes of CE varies, depending upon the suffered organs, and size and grade of the cyst. The metacestodes may invade the various organs (mainly liver and lungs) and cause severe and even fatal clinical outcomes. The occlusion of the pulmonary arterial lumen caused by hydatid disease is rare but life threatening. Therefore, patients with hydatid disease should be carefully evaluated clinically and radiologically to determine whether the hydatid cyst extends into pulmonary vascular structures and heart, leading to pulmonary artery echinococcosis. Here we present a case of pulmonary embolism due to hydatid disease.

Case report A 70-year-old male, who was pathologically diagnosed as having hepatic CE with surgery 22 years ago, and was also diagnosed with a pulmonary hydatid cyst and received surgery 13 years ago. When admitted, the patient complained of an intermittent cough with increasing severity

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Fig. 3 Contrast-enhanced CT shows bilateral pulmonary embolism due to hydatid cyst Fig. 1 Multiple nodules in the right lung field, fan-shaped dense shadow in the left lower lobe overlapped with the heart. Enlargement of the right hilar

Fig. 4 T2-weighted image revealed the cystic nature of the lesions with bright signal intensity and also showed multiple daughter cysts

Fig. 2 Multiple pulmonary hydatid cyst on CT and a cyst in the right upper lobe of the lung containing a freely floating endocyst (the‘‘water lily sign’’)

and hemoptysis for 7 years. He denied fevers, chills, or weight loss. He had been living in the city of Yining located in Xinjiang Uygur Autonomous Region, China, and had no history of travelling to other places. He denied handling livestock or other animals except for contacting dogs during childhood. He accepted cholecystectomy after admission. Serologic testing proved him to be positive for anti-EgCF, anti-EgP, anti-Em2, and anti-EgB antibodies for cystic hydatidosis. No other abnormal laboratory findings were observed. Chest radiographs showed multiple nodules in the right lung field, fan-shaped dense shadow in the left lower lobe overlapped with the heart, apex upturned change, and enlargement of the right hilar (Fig. 1).

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Non-contrast CT (Light-speed Volume CT, GE Medical Systems, USA) revealed the most of multiple cystic masses with low-density (HU values : 8–35), and the larger two lesions, with hypo-attenuating nature, located in the upper lobe (4.7 9 4.6 cm) and right hilar (4.3 9 6.3 cm), which were un-homogeneous and multiple small daughter cysts within the main cyst. A freely floating endocyst (the ‘‘water lily sign’’) was also revealed within a cyst in the right upper lobe of the lung (Fig. 2). We performed dual-source pulmonary CT with contrast injection (iopromide, Ultravist300Ò Schering, Berlin, Germany) to demonstrate the right pulmonary artery and its branches. We saw the left lung lower lobe segmental pulmonary artery as a low-density filling defect in these vessels that resulted in complete occlusion of the lumen (Fig. 3). MRI (Signa Excite HD 1.5T; GE Healthcare, Milwaukee, WI) was attempted, T2weighted and water images revealed the cystic nature of the lesions with bright signal intensity and also showed multiple daughter cysts (Fig. 4). Contrast-enhanced CT and MRI of lung revealed ‘‘ring enhancement sign’’ and wall opacification in some lesion.

Hydatid cyst caused pulmonary embolism

Discussion Echinococcus granulosus (family Taeniidae; genus Echinococcus) is a near-cosmopolitan zoonosis and infects more than three billion people worldwide [2, 3] and has attracted more attention as a major health problem in pastoral regions of the world. At present, the prevalence rates of human cystic hydatidosis range from 0.9 to 2.7 % in China [4]. Hydatid cysts occur mainly in the liver and lungs. The symptoms of hydatid disease generally vary according to the location, size, and grade of the cyst. Lung involvement always occurs after hepatic involvement. Oncospheres are lysed in the proximal digestive tract, releasing six larvae that reach larvae in the liver and pass through sinusoids, suprahepatic veins and the right chambers of the heart or thoracic lymphatic duct, and eventually reside the lungs [5]. The right lung blood and lymphatic flow are slightly more enriched than that in the left lung; therefore, the hydatid cysts in the right lung are more common than that in the left. This case just confirms this view. In addition, oncospheres may reach the lungs through other mechanisms, such as oncospheres pass through the liver without settling. This hypothesis rests on the argument that the hepatic sinusoids are wide enough to let small oncospheres pass [6]. Although oncospheres are smaller than the eggs (22–44 lm), the exact size is not known. The pulmonary artery, pulmonary or systemic embolism caused by hydatid cysts, or vesicles rupturing into the venous or arterial system is an extremely rare complication. In this case, the patient with bilateral pulmonary artery embolism may be due to a venous migration of daughter vesicles to the right heart and then pulmonary arteries. Surgical and autopsy findings indicate that the embolism was caused by vesicles or daughter cysts that act mechanically by obstructing the blood flow, rather than blood clots or added thrombosis. Another mechanism of pulmonary artery embolism might be due to direct extension into or rupture of lung hepatic hydatid lesions near the hilar. Clinically, patients with a small hydatid cyst in the lung usually appear asymptomatic. However, with lesion growth or arising complication(s), patients may have chest pain, coughing, dyspnea, and hemoptysis, as well as allergic reactions [7, 8]. Rupture and infection are frequent complications of pulmonary hydatid disease known as ‘‘complicated cyst.’’ If accompanied with pulmonary embolism, the prognosis of the patient is not good, and he or she may be associated with postoperative morbidity and mortality [7, 9, 10]. Massive pulmonary embolization may result in sudden death and acute cor pulmonale syndromes [11, 12]. Thus, early diagnosis of the pulmonary hydatid embolism is very

important, and may prevent life-threatening embolism via early treatment. It is a challenge for radiology doctors to differentiate between bland pulmonary artery thrombus and hydatid cyst embolism. Hydatid embolism is seen as a lowdensity (cystic) filling defect in the vessel, and membranes can sometimes be detected. In the case presented, daughter cysts of the right pulmonary artery lesion were demonstrated in both CT and MRI. T2-weighted images clearly revealed daughter cysts with bright signal intensity. The imaging properties in combination with clinical history can be helpful to make the differential diagnosis. Although the patient had bilateral pulmonary artery embolism, he did not manifest significant pulmonary hypertension and infarction. This may be due to the dual blood supply system in the lungs. In addition, unlike thrombus, hydatid cyst acting as emboli to embolize the vascular structure(s) might appear softer and grow slowly, so that the patient gradually tolerated this state, and no obvious symptoms manifested. Imaging plays an important role in the diagnosis of hydatidosis. The appearances may vary due to the growth of the parasite and its relationship to adjacent lung tissue. In this case, CT demonstrated complications of lung HC, such as rupture of the cyst into the lung parenchyma resulting in consolidation which surrounded the lesion. It also disclosed the different types and stages of the HC. Type I cyst had hypo-attenuating nature, being oval or round and having well-defined borders, whereas type II had daughter cysts. If the cyst is infected, the wall described as ‘‘ring enhancement sign’’ on contrast-enhanced CT and MRI would be valuable for diagnosis, while contrast enhancement CT would be useful to evaluate involvement of the heart or pulmonary arteries. CT is superior to other imaging modalities in observing intracystic gas, minute calcifications, and common complication of rupture [13– 15]. MRI has unusual imaging modalities to observe the lung hydatid cysts and complication; however it is superior to CT in terms of displaying the daughter cysts and membrane on T2/T1-weighted images. Although chest radiographs are not usually diagnostic modalities, they also provide some information, such as calcifications. Ultrasound is the preferred diagnostic method because of its low cost and availability. However, it is sometimes inadequate in making the initial diagnosis. In addition, contrast-enhanced CT or MRI has the advantage of evaluation of vascular structures to exclude involvement of the heart or pulmonary arteries. Although hydatidosis alone is not difficult to be diagnosed, patients with cough and hemoptysis, chest pain should be carefully evaluated to exclude HC complications deprived from heart and vital vessels. We reported this unusual case to draw attention to diagnose via imaging

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modalities for the pulmonary embolism due to hydatid disease. Moreover, we would like to emphasize that hydatidosis patients with the abnormalities mentioned above need to be examined carefully to figure out the vascular involvement. Acknowledgments The authors thank Zhou Cheng, Nuer Biyemu, and Wang Haitao for help with the images, literature search and examination; Jingxiong Wang and Ashley Benham for their editorial inputs. The authors of this manuscript are responsible for its content. Conflict of interest of interest.

The authors declare that they have no conflict

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5. Sokouti M, Golzari SE, Tizro P, Khanli HM, Ghabili K (2013) Genitourinary hydatid disease. Int Urol Nephrol 45:759 6. Heath DD (1971) The migration of oncospheres of Taenia pisiformis, T.serialisand Echinococcus granulosuswithin the intermediate host. Int J Parasitol 1:145–152 7. Kuzucu A, Soysal O, Ozgel M, Yologlu S (2004) Complicated hydatid cysts of the lung: clinical and therapeutic issues. Ann Thorac Surg 77:1200–1204 8. Khuroo MS (2002) Hydatid disease: current status and recent advances. Ann Saudi Med 22:56–64 9. Turgut AT, Altin L, Topc¸u S, Kilic¸og˘lu B, Aliinok T, Kaptanog˘lu E, Karademir A, Kos¸ ar U (2007) Unusual imaging characteristics of complicated hydatid disease. Eur J Radiol 63:84–93 10. Ozvaran MK, Ersoy Y, Uskul B, Unver E, Yalcin E, Baran R, Morice RC (2004) Pleural complications of pulmonary hydatid disease. Respirology 9:115–119 11. Sirmali M, Gezer S, Yol S, Kaya S (2006) Hydatid cyst of the pulmonary artery secondary to hepatic hydatid cyst embolism. Acta Chir Belg 106:441–442 12. Aribas OK, Kanat F, Turk-Aribas E, Erayman I, Yuksek T (2007) Embolisation of hydatid cysts in the pulmonary artery presenting with haemoptysis. Neth J Med 65:109–111 13. Nemes A, Geleijnse ML, van Geuns RJ, Caliskan K, Michels M, Soliman OI et al (2006) Evaluation of pericardial hydatid cysts by different echocardiographic imaging modalities. Int J Cardiovasc Imaging 22(5):647–651 14. Khuroo MS (2002) Hydatid disease: current status and recent advances. Ann Saudi Med 22:56–64 15. Koul PA, Koul AN, Wahid A (2000) CT in pulmonary hydatid disease, unusual appearances. Chest 118:1645–1647

Rare reason for pulmonary embolism: one case of pulmonary hydatid cyst and review of the literature.

Pulmonary embolism due to hydatid disease has been rare clinically. Here we present a patient case who suffered from multiple cystic hydatidosis withi...
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