Case Report

Percutaneous Transcatheter Embolization of Pulmonary Artery Pseudoaneurysms Causing Massive Hemoptysis: Two Different Case Scenarios

Vascular and Endovascular Surgery 2014, Vol 48(2) 171-175 ª The Author(s) 2013 Reprints and permission: sagepub.com/journalsPermissions.nav DOI: 10.1177/1538574413513339 ves.sagepub.com

Souheil Saddekni, MD, FSIR, FAHA1, Ahmed Kamel Abdel Aal, MD, PhD1, Santhosh Gaddikeri, MD2, Samir Hadi, MD1, and Satinder P. Singh, MD1

Abstract Systemic bronchial arterial circulation is the most common source for massive hemoptysis. Rarely, the source of bleeding can be the pulmonary artery. We report 2 different case scenarios of massive hemoptysis due to different etiologies, in which the source of bleeding was the pulmonary artery. Both the patients were treated with percutaneous transcatheter embolotherapy using coils. The 2 cases highlight the importance of considering pulmonary arterial etiology as the cause of hemoptysis while reporting diagnostic computed tomography studies and tailoring the interventional technique toward performing pulmonary angiography instead of searching for a systemic arterial source for hemoptysis. Keywords embolization, pseudoaneurysm, pulmonary, hemoptysis, percutaneous, transcatheter, embolotherapy, endovascular, minimally invasive

Introduction Massive hemoptysis is commonly defined as expectoration of 300 to 600 mL of blood over a period of 24 hours. However, even a small amount of hemorrhage can be life threatening depending on the ability to maintain the patient’s airway. Thus, a more functional definition of massive hemoptysis is any amount that is sufficiently life threatening and necessitates interventional management. Systemic bronchial arterial circulation is the most common source for massive hemoptysis. Rarely, the source of bleeding can be the pulmonary artery. We report 2 different case scenarios of massive hemoptysis due to different etiologies, in which the source of bleeding was the pulmonary artery. Both patients were treated with percutaneous transcatheter embolotherapy using coils.

Case Reports Case 1 A 60-year-old African American male patient with history of hypertension, rheumatoid arthritis, and chronic obstructive pulmonary disease (COPD) was transferred to our institution from outside hospital with a single episode of massive hemoptysis, which required emergent intubation and transfusion of 6 units of packed red blood cells and 2 units of fresh frozen plasma. Clinical examination of the patient revealed gross deformity

of the hand and feet with ulnar deviation and rheumatoid nodules. Bronchoscopy, cytology wash, culture, fungal stain, and acid-fast stain were all done in the outside hospital and were negative. Multiple repeated induced sputum acid-fast bacillus smears along with purified protein derivative as well as blood and urine cultures were all negative. Chest radiograph demonstrated inhomogeneous air space consolidation in the right upper and both lower lobes. There was no obvious cavitation, mass, adenopathy, or pleural effusion. An emergent computed tomography (CT) angiogram of the chest was done to further evaluate the source of hemoptysis. The CT angiogram together with the postprocessed maximum intensity projection and 3-dimensional volume rendered images that confirmed the presence of right upper lobe consolidation with a small contrast-filled cavity communicating with the posterior subsegmental branch of the right upper

1 Department of Radiology, University of Alabama at Birmingham (UAB), Birmingham, AL, USA 2 University of Washington, Seattle, WA, USA

Corresponding Author: Ahmed Kamel Abdel Aal, Department of Radiology, University of Alabama at Birmingham (UAB), 619 19th Street South, Birmingham, AL 35249, USA. Email: [email protected]

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Figure 1. A 60-year-old African American male patient with history of massive hemoptysis. A, Maximum intensity projection and (B) 3-dimensional volume rendered images showing right upper lobe consolidation (large arrow) with a small contrast-filled cavity (small arrow) communicating with the posterior subsegmental branch of the right upper lobe pulmonary artery (arrowheads) suggesting contained pseudoaneurysm.

Figure 3. Angiogram of the posterior segmental branch of the right upper lobe pulmonary artery demonstrating a small well-defined pseudoaneurysm (small arrow) supplied by a third-order subsegmental branch (arrowhead).

Figure 2. Right pulmonary angiogram demonstrating a small welldefined pseudoaneurysm (small arrow) in the late venous phase in the upper lobe.

lobe pulmonary artery suggesting contained pseudoaneurysm (Figure 1). The patient was then referred to our department for possible endovascular management of his right upper lobe pseudoaneurysm. After gaining access through right femoral vein approach, initial angiogram of the right main pulmonary artery was performed using a 7F-angled pigtail catheter (AP2; Cook, Bloomington, Indiana) which showed a small well-defined pseudoaneurysm that was best seen in the late venous phase (Figure 2). The posterior segmental branch of the right upper lobe pulmonary artery was catheterized with a 7F guide catheter (Envoy; Cordis, Miami, Florida), and angiogram was performed and demonstrated a small well-defined pseudoaneurysm supplied

Figure 4. Follow-up angiogram after placement of multiple variablesized detachable hydrogel coils (arrowheads) in the pseudoaneurysm as well as the feeding artery confirming occlusion of the feeding artery and the pseudoaneurysm.

by a third-order subsegmental branch (Figure 3). The pseudoaneurysm was then catheterized using a 2.8F microcatheter (Progreat; Terumo, Tokyo, Japan). Multiple variable-sized detachable hydrogel coils (Azur; Terumo) were deployed in

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Figure 5. A, Coronal and (B) axial contrast-enhanced computed tomography (CT) scan of the chest demonstrating an area of consolidation containing a large contrast blush (arrowhead) indicating pseudoaneurysm within the left lower lobe with a feeding artery (small arrow) communicating with one of the basal segmental branches of the left pulmonary artery.

Case 2

Figure 6. Left pulmonary angiogram demonstrating pseudoaneurysm (small arrow) in left lower zone in the venous phase.

the pseudoaneurysm as well as the feeding artery. Follow-up angiogram performed through the microcatheter confirmed occlusion of the feeding artery and the pseudoaneurysm (Figure 4). Two days following the procedure, the patient was extubated and the remainder of her hospital stay was uneventful with no recurrent hemoptysis. The patient’s blood parameters continued to improve until she was discharged. In spite of the extensive imaging and laboratory workup for this patient, no definite etiology for this pulmonary artery pseudoaneurysm was identified.

A 68-year-old caucasian male with history of COPD and left lower lobe lung mass was transferred to our institution for management of massive hemoptysis. The patient had hypercapnic respiratory failure with a PCO2 of 100 mm Hg and was intubated. His course was complicated by hypotension and COPD exacerbation and encountered an episode of pulseless electrical activity. After resuscitation, he started having hemoptysis. Bronchoscopy was performed, which demonstrated blood filling the main bronchial arteries bilaterally. A CT scan with contrast was performed, which demonstrated a heterogeneous consolidation with surrounding patchy air space opacities in the dependent portion of left lower lobe. In the periphery of the consolidation, there was a large contrast-filled cavity measuring 24  17 mm2, which appeared to communicate with one of the basal segmental branches of the left pulmonary artery suggesting a pseudoaneurysm (Figure 5). Therefore, the patient was referred to our Interventional Radiology Department for percutaneous transcatheter embolotherapy of his left lower lobe pseudoaneurysm. The patient’s CT findings prompted us to perform pulmonary angiogram before considering bronchial artery embolization. Through a right common femoral vein approach, a left pulmonary angiogram was performed using 7F-angled pigtail catheter (AP2; Cook) placed in the left main pulmonary artery. Left pulmonary angiogram showed faint blush in left lower lung zone in the venous phase images (Figure 6). A coaxial system of a 7F sheath (Flexor Ansel; Cook) placed in the distal left main pulmonary artery and a 4F catheter (KMP; Cook) placed in the inferior left pulmonary artery were used, and a 2.4F microcatheter (Progreat; Terumo) was used to catheterize the basal segmental branch of the left pulmonary artery supplying the pseudoaneurysm. Angiogram was then performed and documented the presence of pseudoaneurysm supplied by this artery (Figure 7). Multiple variablesized pushable coils (Tornado; Cook) were then deployed across the neck and the feeding artery of the pseudoaneurysm.

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Figure 7. Angiogram after catheterization of one of the basal segmental branches of the left pulmonary artery using a microcatheter (arrowheads) documents the presence of a pseudoaneurysm (small arrow) supplied by this artery.

A follow-up angiogram showed occlusion of the embolized feeding artery and the pseudoaneurysm (Figure 8). The patient was eventually extubated 5 days following the procedure and had no further episode of hemoptysis during his hospital stay till he was discharged. Further workup of the patient revealed that the cause of hemoptysis was squamous cell carcinoma of the left lower lobe.

Discussion Massive hemoptysis is commonly defined as expectoration of 300 to 600 mL of blood over a period of 24 hours.1-4 However, even a small amount of hemorrhage can be life threatening depending on the patient’s airway patency. Therefore, a more realistic definition of massive hemoptysis is any amount that is sufficient to cause a life-threatening condition and should be used when deciding whether the patient needs interventional management.4-6 Risk factors for pulmonary pseudoaneurysm formation are tuberculosis,7-9 thoracic trauma, iatrogenic such as peripheral inflation of Swan-Ganz catheters,10 septic emboli, pulmonary abscess, neoplasm, bronchiectasis, Behcet disease,11 and any other acute or chronic inflammation of the lung. The classic Rasmussen aneurysm12 was first reported by Fearn in 194113 and was described as a pseudoaneurysm resulting from the erosion of peripheral pulmonary arterial branches by a tuberculous cavity. Later, it was also reported by Plessinger and Jolly.14 The usual presenting symptom of pulmonary artery pseudoaneurysm is hemoptysis, which is usually massive.9 Bronchoscopy usually allows localization of the pseudoaneurysm to a particular lobe by detecting the source

Figure 8. Follow-up angiogram after deployment of multiple variablesized pushable coils (small arrow) across the neck and the feeding artery of the pseudoaneurysm shows occlusion of the feeding artery and the pseudoaneurysm.

of bleeding. Bronchial arteries are usually evaluated first as a source of hemoptysis, regardless of the etiology. If the bronchial arteriogram is normal, then other systemic arteries such as the intercostal, subclavian, and internal mammary arteries as well as the pulmonary arteries are evaluated. Although pulmonary angiography is considered the gold standard for the evaluation of the pulmonary arterial tree, it has some limitations compared to axial imaging such as CT. Limitations include failure to diagnose-associated extravascular findings like a cavity or a mass, lower contrast resolution, and the need for more selective distal catheterization to demonstrate the pseudoaneurysm in certain cases. For these reasons, a multidetector CT angiography is currently the diagnostic technique of choice.15 There are 2 options for the treatment of pulmonary artery pseudoaneurysm. One is surgery and the other is minimally invasive endovascular embolization. Surgical options could vary depending on the location and consist of any variations such as lobectomy, pneumonectomy, and hilar clamping with direct pulmonary arterial repair or ligation of the pulmonary artery. However, surgery requires a significantly invasive open thoracic procedure with mortality rate as high as 43%.16 Hence, surgery is reserved as the last option for refractory or recurrent pulmonary artery pseudoaneurysms if endovascular treatment failed or was not feasible. Endovascular therapy is performed by embolization of the pseudoaneurysm and/or the feeding artery using a variety of embolic agents such as steel or platinum coils, glue, detachable balloons, Amplatzer Vascular plugs (St Jude Medical, Plymouth, Massachusetts), or covered stents. The choice of the embolic agents or covered stents depends on the number, size, location,

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and width of the neck of the pseudoaneurysm. Complications of the endovascular therapy may be pulmonary arterial dissection due to catheter manipulation, pulmonary infarction due to nontarget embolization, and rarely renal failure due to iodinated contrast used during the procedure.17,18

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Conclusion In conclusion, hemoptysis is commonly caused by inflammatory disease of the lung parenchyma with systemic arterial supply originating from bronchial and other systemic arteries. Occasionally bleeding may arise from pulmonary arteries. We report here 2 cases of pseudoaneurysms resulting from erosions of segmental pulmonary artery branches that essentially could be called Rasmussen pseudoaneurysms. The 2 cases highlight the importance of considering pulmonary arterial etiology as the cause of hemoptysis while reporting diagnostic CT studies and tailoring the interventional technique toward performing pulmonary angiography instead of searching for a systemic arterial source for hemoptysis. Multidetector CT angiography of the chest may facilitate early and accurate detection of the source of hemoptysis and guides minimally invasive endovascular treatment.

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Declaration of Conflicting Interests

12.

The author(s) declared no potential conflicts of interest with respect to the research, authorship, and/or publication of this article.

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Funding

14.

The author(s) received no financial support for the research, authorship, and/or publication of this article. 15.

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Percutaneous transcatheter embolization of pulmonary artery pseudoaneurysms causing massive hemoptysis: two different case scenarios.

Systemic bronchial arterial circulation is the most common source for massive hemoptysis. Rarely, the source of bleeding can be the pulmonary artery. ...
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