INTRODUCTION:A 9- year- old girl with a history of chronic cough diagnosed as asthma presented with acute hemoptysis. Investigations revealed an esophageal bronchus.
CASE PRESENTATION:The patient coughed up fresh blood while sitting at school on three occasions in two weeks. A tickle in her throat and mild chest pain preceded the onset of hemoptysis. She had no bleeding elsewhere. She had lost four pounds over two months. There was no history of fever, night sweats or TB contact. She had been diagnosed with asthma four years previously, for which she used daily inhaled steroid and prn albuterol twice weekly. Her cough was usually aggravated by running and eating. On examination, her vital signs were stable. Fine crepitations were heard in the right lower lung field. Her fingers were clubbed. Chest X-ray revealed ill-defined opacities with patchy infiltration around the hilum and in the superior segment of the right lower lobe. The coagulogram, immunology workup and tuberculin skin test were all negative. CT scan of the chest revealed a communication between the lower esophagus and 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 an extra opening in the second order bronchus of the superior segment of the right lower lobe with whitish mucus plugging, but no active bleeding or endobronchial lesion. Gastroscopy revealed a large abnormal connection to the lung from the distal esophagus, but no esophagitis, gastritis or peptic ulcer. Bronchoalveolar lavage cultures grew Candida albicans. A lobectomy 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 the superior segment bronchi, along with bronchopneumonia, lymphocytic bronchiolitis, and intra-alveolar hemorrhage. Thickened irregular islands of cartilage with respiratory and gastrointestinal epithelium colocalized in the connection, suggesting a congenital bronchopulmonary foregut malformation, most likely esophageal bronchus. Food and Actinomycetes particles were found in bronchioles and lung parenchyma, suggesting aspiration pneumonia.
DISCUSSIONS:A communicating bronchopulmonary foregut malformation is a rare condition. Repetitive aspiration of food particles leads to bronchiolitis, bronchopneumonia and bronchiectasis that can present with postprandial cough, hemoptysis, failure to thrive and digital clubbing. Some cases without esophageal atresia may not be recognized until mid childhood or even adulthood. Esophagography is the most useful diagnostic procedure. The abnormal communication is usually more easily visualized by esophagoscopy than bronchoscopy.
CONCLUSION:This case serves to remind clinicians that patients with cough and wheeze unresponsive to asthma therapy require further investigations, especially in the presence of failure to thrive or digital clubbing.
DISCLOSURE:Harutai Kamalaporn, No Financial Disclosure Information; No Product/Research Disclosure Information