AN ESOPHAGEAL BRONCHUS IN A 9-YEAR-OLD GIRL PREVIOUSLY DIAGNOSED WITH COUGH-VARIANT ASTHMA

Abstract

The patient coughed up fresh blood whilesitting at school on three occasions in two weeks. A tickle in her throat andmild chest pain preceded the onset of hemoptysis. She had no bleedingelsewhere. She had lost four pounds over two months. There was nohistory of fever, night sweats or TB contact. She had been diagnosed withasthma four years previously, for which she used daily inhaled steroid andprn albuterol twice weekly. Her cough was usually aggravated by runningand eating. On examination, her vital signs were stable. Fine crepitationswere heard in the right lower lung field. Her fingers were clubbed. ChestX-ray revealed ill-defined opacities with patchy infiltration around thehilum and in the superior segment of the right lower lobe. The coagulo-gram, immunology workup and tuberculin skin test were all negative. CTscan of the chest revealed a communication between the lower esophagusand the superior segment of the right lower lobe of 9 mm in diameter.There was right lower lobe consolidation without any abnormal vessels.Oral contrast material freely entered the lung. Bronchoscopy revealed anextra opening in the second order bronchus of the superior segment of theright lower lobe with whitish mucus plugging, but no active bleeding orendobronchial lesion. Gastroscopy revealed a large abnormal connectionto the lung from the distal esophagus, but no esophagitis, gastritis orpeptic ulcer. Bronchoalveolar lavage cultures grew Candida albicans. Alobectomy and esophageal repair were performed without complication.Pneumonia was treated with cefuroxime and clarithromycin. At pathology,there were bronchiectatic changes of the right lower lobe, especially thesuperior segment bronchi, along with bronchopneumonia, lymphocyticbronchiolitis, and intra-alveolar hemorrhage. Thickened irregular islandsof cartilage with respiratory and gastrointestinal epithelium colocalized inthe connection, suggesting a congenital bronchopulmonary foregut mal-formation, most likely esophageal bronchus. Food and Actinomycetesparticles were found in bronchioles and lung parenchyma, suggestingaspiration pneumonia.