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tapraid4/jvi-jvir/jvi-jvir/jvi99908/jvi2998d08z patankara S⫽1 9/22/09 11:42 Art: 1169
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Endovascular Embolization of Pulmonary Sequestration in an Adult From: K. S. Madhusudhan, MD, Chandan J. Das, MD, R. Dutta, Arvind Kumar, MS, Ashu S. Bhalla, MD Departments of Radiology and Surgery (R.D., A.K.) All India Institute of Medical Sciences Ansari Nagar New Delhi 110029, India Editor: Pulmonary sequestration is a congenital malformation representing a mass of nonfunctional pulmonary tissue that does not communicate with the bronchial tree or the pulmonary arteries (1). Two forms of pulmonary sequestration have been described. The intralobar variety is commonly seen in young adults, who often presents with recurrent pneumonia, whereas the extralobar type is commonly seen in the pediatric age group (1). Hemoptysis is uncommon and may range from intermittent to massive life-threatening episodes. Conventionally, pulmonary sequestration is treated surgically. Several studies in the literature describe embolization as a safe alternative to surgery, especially in infants (2– 4). However, very few reports of embolization of sequestration in adults are found in the English literature (5). We describe a case of pulmonary sequestration in an adult presenting with hemoptysis who was treated with endovascular embolization. A 31-year-old man presented to the surgery department with a 2-month history of recurrent hemoptysis. The amount of hemoptysis was in the range of 25–50 mL/d and was bright red in color. There was no history of fever. No past or present history of pulmonary tuberculosis was present. Clinical examination findings were unremarkable. Chest radiograph showed a small patch of consolidation in the right lower zone. The patient was given a course of antibiotics (amoxicillin plus clavulanate for 7 days). Repeat chest radiograph after 4 weeks did not show any change in the appearance of the consolidation. Contrast-enhanced computed tomography (CT) of the chest subsequently showed an area of consolidation in the posterior basal segment of the right lower lobe. A tortuous feeding artery was seen arising from the abdominal aorta. Fiberoptic bronchoscopy was done and showed normal findings. The patient underwent CT angiography, with an imaging diagnosis of pulmonary sequestration. It showed a prominent arterial feeder vessel arising from the lower thoracic aorta supplying the patch of consolidation (Figure, a). Venous drainage was seen into the left atrium via the inferior pulmonary veins. Based on these findings, a diagnosis of intralobar sequestration was made. As there was no other lesion on CT scan and the bronchoscopy findings were normal, we presumed that sequestration was the cause of hemoptysis. As the patient refused surgery, we decided to perform transarterial embolization after obtaining informed consent.
None of the authors have identified a conflict of interest. DOI: 10.1016/j.jvir.2009.09.002
Digital subtraction angiography was done through a right transfemoral route. A flush aortogram initially performed with a 5-F pigtail catheter showed the abnormal branch from the descending thoracic aorta, but selective angiography of the feeding artery with a 5-F Picard catheter showed multiple smaller branches in addition to the larger branch supplying the sequestration (Figure, b and c). There was no element of arteriovenous fistula as the pulmonary veins opacified after capillary blush and not with the artery. Polyvinyl alcohol particles (PVA; 500 –700 m; Cook, Bloomington, Indiana) were used for embolization. Particles of this size would not cross the capillaries and hence the risk of systemic embolization could be avoided. This was followed by embolization with two stainless-steel coils (Cook). After embolization, digital subtraction angiography showed no abnormal blush, although a few small branches were seen (Figure, d). No complications were encountered and the patient was discharged the next day. Hemoptysis resolved completely after embolization. The patient remains asymptomatic 1 year after the procedure and is participating in regular telephone follow-up. Intralobar sequestration lies within the pleura of the normal lung and is the more common of the two types of pulmonary sequestration. It is commonly seen in the lower lobes and receives systemic blood supply from the thoracic or abdominal aorta or its branches. Venous drainage usually occurs to the pulmonary veins (4). Although a left-to-left shunt occurs in pulmonary sequestration, no definite microscopic or macroscopic arteriovenous fistula is seen. Arteriovenous malformation occurring in the lung is a different entity, although some consider it as a variant of pulmonary sequestration (6). Hemoptysis in pulmonary sequestration occurs as a result of increased systolic and diastolic pressure of systemic arteries supplying the anomalous tissue, thus increasing the capillary pressure. Hemoptysis is usually minor, and is often associated with pneumonia (1). Imaging plays an important role in the evaluation of pulmonary sequestration. Demonstration of systemic supply to the abnormal tissue is important for making the diagnosis. CT angiography or magnetic resonance angiography is effective in demonstrating the major systemic feeder vessel and providing anatomic information for planning surgery (7). Digital subtraction angiography may be rarely required for diagnosis (7). A normal capillary phase is seen in pulmonary sequestration unlike in arteriovenous malformation, in which capillaries are abnormal, resulting in direct fistulous communication between arteries and veins (6). The conventional treatment for pulmonary sequestration has been surgical removal of the abnormal tissue or lobectomy, which is associated with morbidity and complications (1,2). Also, surgical excision in asymptomatic patients has been debated in view of reports of spontaneous regression of pulmonary sequestration in children (8). Embolization is an alternative technique in the treatment of symptomatic sequestration. However, all the relevant studies of which we are aware were been performed in infants and children (2– 4). Embolization of sequestration in adults has been sporadically documented in the English literature (5). It is thought that embolization causes thrombosis of the feeding artery, which results in reduced perfusion, loss of vascularity, and progressive infarction of the anomalous lung tissue, which finally becomes fibrosed. This leads to reduction in the size of the sequestration and some-
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Figure. (a) Volume-rendered image of CT angiogram shows the prominent feeder vessel arising from the descending thoracic aorta (arrowhead) with venous drainage into the left atrium through inferior pulmonary vein (arrow). (b) Selective angiogram of the feeding artery shows abnormal parenchymal blush in right lower lobe (arrows). (c) The venous drainage is seen into left atrium via inferior pulmonary veins (arrow). (d) Postembolization angiogram shows coils in the feeding artery with obliteration of the parenchymal blush and absence of venous filling.
times to its involution (2). This effect is equivalent to that of surgical excision of the tissue. Endovascular treatment is less invasive than thoracotomy and has a lower incidence of complications. Metallic coils represent the preferred embolization material in most of the reported cases (4). Other materials used are PVA particles, microcoils, n-butyl cyanoacrylate, absolute alcohol, and gelatin sponge particles. Lee et al (5) preferred gelatin sponge particles and microcoils in view of the risk of regurgitation of permanent embolic materials, which would cause rapid and massive tissue necrosis. A combination of coils and PVA has also been used (4). We too preferred to use PVA particles initially to embolize the smaller branches, in which coil embolization would have been difficult. This also slowed the flow in the major feeding artery and allowed the coil to stay in a proximal location. The risk of nontarget embolization should be considered, especially if preliminary digital subtraction angiography shows an element of arteriovenous fistula. The pulmonary veins in our case were opacified in the venous phase, and hence we presumed there was no major shunting. Nevertheless, presence of minute shunts could not be excluded, so we used larger PVA particles to avoid systemic embolization. Partial
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embolization and nonregression of the sequestered tissue after embolization occurs in 25%–47% of cases, some of which would later require surgery (3). Various reported complications include distant migration of embolization material causing inadvertent embolization of nontarget arteries, infection, puncture site thrombosis, pain, and fever (3). After reviewing various reports, we suggest the use of coils as embolization material; PVA can be used if the element of shunting is excluded. In conclusion, intralobar sequestration in adults may present with hemoptysis. Although surgery is the conventional choice of treatment, embolization of the systemic arteries can be a safe alternative method. Metallic coils are most commonly used. More studies are needed to firmly establish its role in adults. References 1. Frazier AA, Rosado de Christenson ML, Stocker JT, Temleton PA. Intralobar sequestration: radiologic pathologic correlation. Radiographics 1997; 17:725–745. 2. Lee KH, Sung KB, Yoon HK, et al. Transcatheter arterial embolization of pulmonary sequestration in neonates: long-term follow-up results. J Vasc Interv Radiol 2003; 14:363–367.
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3. Lee BS, Kim JT, Kim EA, et al. Neonatal pulmonary sequestration: clinical experience with transumbilical arterial embolization. Pediatr Pulmonol 2008; 43:404 – 413. 4. Tokel K, Boyvat F, Varan B. Coil embolization of pulmonary sequestration in two infants: a safe alternative to surgery. AJR Am J Roentgenol 2000; 175:993–995. 5. Muñoz JJ, García JA, Bentabol M, Padín MI, Serrano F. Endovascular treatment of hemoptysis by abnormal systemic pulmonary artery supply. Cardiovasc Intervent Radiol 2008; 31: 427– 430.
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6. Wang HC, Kuo PH, Liaw YS, et al. Diagnosis of pulmonary arteriovenous malformations by colour Doppler ultrasound and amplitude ultrasound angiography. Thorax 1998; 53:372–376. 7. Abbey P, Das CJ, Pangtey G, Seith A, Dutta R, Kumar A. Imaging in bronchopulmonary sequestration. J Med Imaging Radiat Oncol 2009; 53:22–31. 8. Pena PG, Lucaya J, Hendry GMA, McAndrew PT, Duran C. Spontaneous involution of pulmonary sequestration in children: a report of two cases and review of the literature. Pediatr Radiol 1998; 28:266 –270.
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