Thoracic endovascular aortic repair and coil embolization of the pulmonary artery for primary racemose hemangioma of the bronchial artery with a bronchial-pulmonary artery fistula

Abstract

A 69-year-old male visited a doctor with dyspnea. A bronchial-pulmonary artery fistula, which is called racemose hemangioma, and dilated pulmonary artery were detected by a computed tomography (CT) scan. The bronchial-pulmonary artery fistula can cause lethal hemoptysis, therefore, we performed thoracic endovascular aortic repair (TEVAR) to seal the bronchial artery and coil embolization of the pulmonary artery. Postoperative CT showed the thrombosed racemose hemangioma of the bronchial artery. TEVAR and coil embolization of the pulmonary artery is considered to be a useful treatment option for a racemose hemangioma.

Embolization of a Bronchial Artery–Pulmonary Artery Arterio–Venous Malformation

“…While the most common presentation of BP-AVM is that of hemoptysis, 2–4) our case presented with exertional dyspnea which markedly improved following embolization. We are not fully certain of the etiology but postulate that it could be due to resolution of pulmonary hypertension and congestion with embolization of the AVM.…”

“…Surgery (lobectomy) remains the main treatment BP-AVM with bronchial artery embolization being described as a potential alternative. 3,4) From the perspective of embolotherapy, BP-AVM differs from the commonly seen pulmonary AVM. BP-AVM represents a systemic artery to pulmonary artery shunt, that is; a left to right shunt versus a right to left shunt, and therefore has a much higher shunt speed.…”

“…Within the literature, success with bronchial artery embolization in BP-AVM remain mixed due to delayed vascular recanalization and concern for pulmonary embolism in the high-flow condition. 3,4) More importantly, the authors in the described cases performed solely arterial side embolization without embolization of the outflow. Based on this, we chose to first embolize the outflow pulmonary artery to reduce the shunting rate (left-to-right shunt physiology) which not only reduces the risk of pulmonary embolism but also makes subsequent arterial side embolization easier.…”

“…Our “bi-directional” embolization approach deviates from the described “bronchial artery embolization only approach” and is further supported by another case in the literature which pulmonary artery embolization (outflow) was combined with thoracic endografting (inflow) in a case of BP-AVM. 4)…”

A bronchopulmonary artery fistula in a horse

“…In humans, BPAF are classified into congenital and acquired types, according to their causes [6,7]. Congenital BPAF are caused by developmental vascular anomalies that arise during the fetal period [14].…”