Pulmonary Artery-Tracheal Fistula After Coil Implantation for Behcet’s Disease

Abstract

Behcet’s disease (BD) is a rare autoimmune disorder that results in diffuse full-thickness vasculitis. Pulmonary artery aneurysms (PAAs) and hemoptysis are known complications of this disease process, with high morbidity and mortality for affected patients. Although medical, endovascular, and surgical treatment strategies have all been described in the literature, there are little data to describe the long-term outcomes of these various treatment modalities and there continues to be a lack of clearly defined algorithms for the management of these patients. We report a case of PAA in the setting of BD who was treated over the course of many years with medical therapy and coil embolization but ultimately failed treatment, sustained a complication of coil erosion and migration into the trachea twice, and required surgical lobectomy for definitive management. We discuss an algorithm for the management of patients with BD who have PAAs.

Embolization of pulmonary artery aneurysms in a patient with Behçet’s disease complicated by coil erosion into the airway

“…5 Coil erosion and migration into the mainstem bronchus is a rare complication of coil embolization with an unknown incidence, described in a case report occurring 2 years after embolization for which a left lower lobectomy with resection of the PAA and coil was performed. 6 Our case differs from this previously published case report given the short time between embolization and symptom onset. Bleeding risk following bronchoscopy to remove migrated endovascular coils is not known, but one series of 1035 bronchoscopies in J o u r n a l P r e -p r o o f 6 children to remove aspirated foreign bodies reported only 6 cases of bleeding from the tracheobronchial system.…”

Bronchoscopic Management of Hemoptysis Caused by Airway Erosion From Vascular Coils

“…Erosion and migration of coils remain extremely rare complications, with few reported cases in the literature, and none reported among patients with HSS to our knowledge 3–5 . Management has typically been conservative, although 1 patient with Behcet disease was reported to ultimately require surgical resection due to repeated vascular coil erosion and formation of a pulmonary arterial-tracheal fistula 3,5 . Specific treatment of coil migration into the airways remains largely dependent on the patient’s preferences, comorbidities, and available clinical expertise.…”

Endovascular Plug for Endobronchial Management of an Expectorated Pulmonary Artery Embolization Coil: A Case Report

“…7 In addition, there is a report of a patient with Behcet disease who had recurrent hemoptysis and transbronchial migration of aneurysm coils twice, ultimately requiring lobectomy. 8 Finally, there are case reports of embolization coils placed for arteriovenous malformations resulting in transbronchial migration years later, often requiring resection of affected segments. 9 Only one of these cases involved clipping and removing the extruded coil via bronchoscopy.…”