Resection of infected bulla associated with isolated unilateral absence of pulmonary artery: a case report

Abstract

BackgroundA unilateral absence of the pulmonary artery (UAPA) is a rare congenital anomaly, which in most patients is associated with congenital cardiovascular abnormalities. Some patients with UAPAs do not have associated cardiovascular abnormalities, called isolated UAPA. Patients with isolated UAPA generally have a mild clinical course and may not be diagnosed until adulthood, but repeated complications such as infections may make surgery difficult.Case presentationA 34-year-old female patient with isolated right UAPA was referred to our hospital with a complaint of fever. Chest radiography and computed tomography showed an infectious cavity in the right upper lung. Surgery was scheduled for the patient; however, her preoperative condition needed to be optimized. She was evaluated for pulmonary hypertension and the collateral arteries were mapped out, and a waiting period after treatment of infection was set to avoid perioperative intrathoracic infections. Resection of the infected bulla was performed without any complications. No recurrence of infection was observed two years post-operatively. The residual lung expanded enough to fill the dead intrathoracic space.ConclusionsThis report suggests the importance of adequate preoperative evaluation and perioperative management for patients with isolated UAPA who require surgical resection.