Abstract
Systemic artery-to-pulmonary vessel fistula (SAPVF) is a rare vascular anomaly, with acquired forms typically arising secondary to intrathoracic inflammation, infection, trauma, or thoracic surgery. We report a case of a 21-year-old man with a history of bilateral video-assisted thoracoscopic surgery (VATS) bullectomy for primary spontaneous pneumothorax who developed bilateral SAPVF. Contrast-enhanced computed tomography revealed abnormally dilated pulmonary vessels with systemic arterial communications in both lungs, corresponding to previous surgical incision sites. On the left side, SAPVF developed despite the surgical wound separated from the lung by oxidized regenerated cellulose sheets. Due to multiple systemic feeding arteries, embolization was considered ineffective, and surgery was avoided owing to the high risk of postoperative reformation. The patient has been managed conservatively, with no progression observed over a two-year follow-up period. This case highlights that SAPVF can occur even after minimally invasive surgery such as VATS, emphasizing the need for careful follow-up and further evaluation of optimal preventive strategies.