Secondary systemic artery to pulmonary artery and pulmonary vein fistulas following the video-assisted thoracic surgery for pneumothorax: a case report

BACKGROUND: The systemic artery to pulmonary vessel fistula (SAPVF) is a vascular anomaly characterized by penetration of nonbronchial systemic chest wall arteries into the lung parenchyma. To our knowledge, about 150 cases of SAPVF have been reported to date. Fifteen percent of SAPVF are congenital and occur in the presence of cardiopathy or pulmonary artery hypoplasia. Secondary SAPVF are caused by pleural adhesions that occur subsequent to inflammatory changes associated with conditions such as pleuritis, empyema, trauma, and surgery. Though several cases of secondary SAPVF as a post coronary artery bypass graft (CABG) complication have been reported, secondary SAPVF especially following video-assisted thoracic surgery (VATS) are relatively rare. CASE PRESENTATION: A 19-year-old man was admitted to our hospital because of recurrence of left pneumothorax. His previous history included left and right pneumothorax at the ages of 15 and 16 years, respectively, which were treated by VATS. VATS was planned for the surgical indication of second postoperative recurrence. In the operation, the lingular segment with dilated pulsating pulmonary vessels adhered to the port scar of the chest wall, which was made at first VATS for pneumothorax. The computed tomography showed an abnormal connection between the branch of the systemic artery of the chest wall and the dilated pulmonary artery and pulmonary vein in the lingular segment. Left subclavian selective arteriography also showed hypertrophic blood vessels arose from the internal thoracic artery, the lateral thoracic artery, and the subscapular artery, which drained into the both the pulmonary artery and the pulmonary vein in the lingular segment. Despite of four sessions of embolization for aberrant arteries, the abnormal blood flow persisted. Partial resection of the left lingular segment was therefore performed. The patient has been disease-free about SAPVF for 2 years and 2 months after the last operation. CONCLUSIONS: We described our experience with a case of secondary SAPVF that was associated with fistulas between a systemic artery and both the pulmonary artery and pulmonary vein, which was developed after first VATS for pneumothorax. Radical resection was safely performed and effective after four sessions of embolization.