An Unusual Complication of Long-term Endobronchial Valves Placed for Persistent Air Leak
Endobronchial valves (EBVs), originally designed for bronchoscopic lung volume reduction, have been increasingly utilized for the management of bronchopleural fistula (BPF) and alveolar-pleural fistula with persistent air leak (PAL) and are currently approved by the Food and Drug Administration on a humanitarian device exemption for the management of postoperative BPF.1–4 EBVs have been associated with a variety of complications including exacerbation of chronic obstructive pulmonary disease, pneumonia, pneumothorax, prolonged air leak, and EBV migration.2–4
A 22-year-old male was referred to the Interventional Pulmonology clinic after 2 previously-placed EBVs were discovered to have migrated into the pleural space. The patient had a complicated medical history consisting of diffuse large B-cell lymphoma diagnosed 5 years before presentation, originally treated with chemotherapy and mediastinal radiation. The following year, a cavitary lesion was identified in his left upper lobe on surveillance chest computed tomography. A left upper lobectomy was performed for a presumed aspergilloma; surgical pathology instead revealed a locus of disease recurrence. After lobectomy, the patient was noted to have a BPF with PAL that persisted despite conservative management. Three EBVs (Spiration Inc., Redmond, WA) were placed in various left-sided segments, with resolution of the air leak and chest tube removal within 4 days. In the 2 years following his surgery, he experienced recurrent episodes of Pseudomonas aeruginosa pneumonia requiring intravenous antibiotics, often with recurrence of infectious and constitutional symptoms at the conclusion of each course of treatment.
At the time of presentation to our institution, he was at his baseline level of dyspnea on exertion and reported occasional small-volume hemoptysis as well as significant weight loss in the previous 6 months. A chest computed tomography revealed a loculated pneumothorax in the former location of the left upper lobe and diffuse pleural thickening. Part of the left upper lobe stump was contiguous with the pneumothorax, representing a large BPF. One EBV remained well positioned in a segmental bronchus, whereas the other 2 appeared to have migrated into the pleural space (Fig. 1). The patient was referred to Interventional Pulmonology for consideration of pleuroscopy and bronchoscopy to obtain microbiological samples and evaluate the feasibility of valve removal, which were suspected to represent niduses for recurrent infection.
Bronchoscopic evaluation was performed to evaluate airway anatomy using a flexible videobronchoscope (Olympus BF Q180) with 5.1-mm outer diameter. Airway survey revealed normal anatomy on the right. On the left, a large BPF was evident in the region of the left upper lobe stump, with a smaller BPF immediately superior it (Fig. 2). One EBV, covered by thick purulent material and granulation tissue, was identified in a left-sided airway and removed uneventfully with flexible forceps (Fig. 2). A videobronchoscope with 4-mm outer diameter (Olympus P190) was then used to pass through the BPF into the pleural space. Within the pleural space, the second EBV was easily identified and removed without incident. Further visual inspection did not reveal the location of the third EBV. Using fluoroscopy, it was found to be embedded in dense pleural adhesions in the anterior cardiophrenic cul-de-sac and extraction was not attempted. The patient tolerated the procedure well with no complications. There was no evidence of disease recurrence or pleural space infection. Cultures of the 2 retrieved EBVs yielded Pseudomonas aeruginosa.
Valve migration is a known complication of EBV placement. We report a unique case in which 2 EBVs migrated through a BPF into the pleural space; to our knowledge, this is the first report of intrapleural EBV migration. Moreover, this novel presentation highlights the potential of long-term EBVs to act as niduses for persistent infection with resistant organisms.