Embolization of Multiple Systemic Artery to Pulmonary Artery Fistula with Recurrent Hemoptysis
A 51-year-old man was admitted to our hospital with recently developed hemoptysis. He was previously healthy and did not have any family history of hemoptysis. He had recurrent hemoptysis for three days and the amount was about 200 mL/day, but no cough, sputum, dyspnea, nor fever. The patient had a history of smoking with more than 30 pack-years. Upon examination, there were no crackles or wheezing suggesting airway diseases, and no cardiac murmurs. Laboratory data showed normal range of hemoglobin (15.8 g/dL), platelet (244,000/mm3), C-reactive protein (1.0 mg/dL), prothrombin international normalized ratio (1.0).\nA chest radiograph revealed increased left parahilar opacity. For further evaluation, he underwent thin-section computed tomography (CT) scan (1.3 mm thickness). The arterial phase CT scan revealed about a 2.5 cm-sized well-enhanced elongated vascular mass abutting on the pericardium in the left upper lobe and an abnormally prominent peripheral pulmonary artery accompanying early enhancement of the left main pulmonary artery (). When the arteries around the lesion were traced, these originated from the mediastinal branch of the left internal mammary artery and the proximal portion of the left common carotid artery. On the lung window setting images, ground glass opacity suggesting aspirated blood was noticed around the lesion (). Based on the CT finding that the lesion had an arterial enhanced vascular mass with early enhancement of the pulmonary artery, this lesion was suggestive of a systemic artery to pulmonary artery fistula.\nSince the patient showed persistent hemoptysis, embolization of the supplying vessels was planned. Prior to the embolization of the causative vessels of the hemoptysis, diagnostic aortography, left subclavian arteriography, and left internal mammary arteriography including selective arteriographies of the branches that were directly supplying the vascular mass were performed. During the diagnostic aortography, 2 feeding vessels from left internal mammary artery to vascular mass were identified (), and coil embolization was performed with a 5-Fr catheter and coaxial 2.0-Fr microcatheter (Progreat; Terumo, Tokyo, Japan) (). Microcoils (Tornado embolization microcoil; Cook, Bloomington, IN, USA) were used. From the angiographic findings, multiple systemic arteries supplied the vascular mass lesion and the contrast material drained early to the left pulmonary artery through the fistula in accordance with the CT findings.\nDuring the left subclavian arteriography, the patient underwent a sudden-onset headache due to an unexpected embolic event at the left superior cerebellum through the left vertebral artery. However, after conservative treatment, the headache subsided and patient did not show any neurological deficits or sequelae.\nBut, patient complained recurrent hemoptysis, so additional diagnostic angiography was performed for searching residual fistulas. In the left subclavian and left internal mammary arteriography, feeding vessels from proximal portion of left subclavian artery were identified (), and residual fistulas from left internal mammary artery were discovered (). And then, coil embolizations to fistulas were performed respectively and the disappearance of them was confirmed ().\nDuring the diagnostic aortography, left bronchial artery () and pericardiacophrenic branch of the left inferior phrenic arteries () were identified as additional supplying branches. For embolization of the left bronchial artery and pericardiacophrenic branch of the left inferior phrenic artery, glue (N-butyl-2-cyanoacrylate) and gelfoam sponge particles were used because these arteries were too small to insert coils ().\nAfter the embolization, the patient showed an intermittent old blood clot like sputum due to aspirated blood for several days without any evident hemoptysis. The patient was followed up for 16 months after embolization. During the follow-up period, the patient had no recurrence of hemoptysis.