Institutional members access full text with Ovid®

Bronchial Atresia With Mucoceles and Concurrent Sinus Mucoceles: First Reported Case and a Review of the Literature

Pamer, Mark J. DO; Lenhardt, Richard MD

doi: 10.1097/CPM.0b013e3181671a5c
Images in Pulmonary Medicine

Bronchial atresia is a rare congenital anomaly of the airways associated with regional airway hyperinflation and bronchial mucocele formation. It is often found incidentally during chest radiography performed in young adults. Most patients are asymptomatic but some report dyspnea or frequent respiratory infections. Management is conservative; there are few indications for surgical intervention. Sinus mucoceles are benign cyst-like structures in the paranasal sinuses that may develop as a consequence of sinus obstruction. The resulting fluid accumulation in the sinuses may eventually cause bony erosion, allowing access to the intracranial or orbital spaces and causing symptoms. Sinus mucoceles are diagnosed by sinus computed tomography and managed surgically or endoscopically.

We present a 43-year-old woman who underwent emergency surgery for acute blindness due to extensive sinus mucoceles. Postoperative chest imaging revealed right upper lobe mucoceles surrounded by hyperlucent lung, suggestive of bronchial atresia. Bronchoscopy confirmed right upper lobe posterior segment bronchial atresia. To our knowledge, this is the first reported case of concurrent sinus mucoceles and bronchial atresia with mucoceles. Their concomitant presence may be coincidental or it may be due to an as-yet-unknown embryological defect of the sinus and bronchial buds of the alimentary tract.

Bronchial atresia is a rare and interesting condition. It has never been associated with concurrent sinus mucocele formation. To our knowledge, we present the first report of the simultaneous presence of both entities in a woman presenting with acute blindness who was subsequently discovered to have abnormal chest radiography. A review of the literature for both conditions follows.

From the Department of Pulmonary and Critical Care Medicine, Rush University Medical Center, Chicago, Illinois.

Address correspondence to: Dr. Mark J. Pamer, DO, Department of Pulmonary and Critical Care Medicine, Rush University Medical Center, 1725 West Harrison Street, Suite 054, Chicago, IL. E-mail: MarkJPamerDO@aol.com.

© 2008 Lippincott Williams & Wilkins, Inc.