INFECTED SIMULTANEOUS MEDIASTINAL BRONCHOGENIC CYST AND ESOPHAGEAL DUPLICATION CYST MASQUERADING AS A LUNG ABSCESS

Abstract

TOPIC: Genetic and Developmental Disorders TYPE: Fellow Case Reports INTRODUCTION: Bronchogenic and esophageal duplication cysts are rare but significant developmental anomalies. We present a case with both types of cyst in a single mass mimicking a lung abscess with a protracted treatment course. CASE PRESENTATION: A healthy 26-year-old female was admitted with onset of left sided pleuritic chest pain and fever over 24 hours. Her exam was remarkable for fever and tachycardia; she had elevated markers of infection. Computed tomography (CT) of chest revealed a well-demarcated cystic mass in the left hemithorax with an air-fluid level. She had no prior chest imaging and denied aspiration events or substance use. She was thought to have an intraparenchymal lung abscess, and underwent CT guided aspiration with the drainage of frank purulent material. Because of suspected intraparenchymal location no drain was placed. Fluid cultures and cytology were negative but her sputum grew mucoid pseudomonas aeruginosa, which was an atypical pathogen in this patient. She was treated with four weeks of intravenous cefepime, and two weeks of levofloxacin and metronidazole, with serial imaging. Pleuritic chest pain persisted despite regression of the lesion over a 10-month period (Figures 1-3); after which she underwent resection through a left video-assisted thoracic surgical approach. Intraoperatively, a large densely adherent inflammatory lesion was connected to the esophagus without intraluminal connection. Thick purulent fluid from the cyst was sent for culture and cytology, which were again negative. Pathology of the cystic mass was consistent with an esophageal duplication cyst (EC) (Figure 4B) with an additional noncommunicating bronchogenic cyst (BC) (Figure 4A). At 6months follow-up visit, she was doing well and free of symptoms. DISCUSSION: To our knowledge this is the first case report of de-novo infected dual BC/EC in an adult. Our case highlights the need for consideration of congenital cysts while working up lung abscesses. The diagnosis may be difficult without surgical resection, as characteristic imaging findings are missing in up to half of cases. 1There is likely a role for aspiration of the cyst for both diagnostic and therapeutic purposes, however efficacy and safety data are limited.2 The optimal timing of cyst resection is also not clear, especially in asymptomatic cysts. There is a minimal evidence to support universal surgical resection, though infected and symptomatic cysts will continue to need surgical intervention. 2,3 CONCLUSIONS: Infected congenital cysts can present a diagnostic and therapeutic challenge. Atypical presentations, and lack of improvement with appropriate therapy, are important clues to the diagnosis. REFERENCE #1: St-Georges R, Deslauriers J, Duranceau A, Vaillancourt R, Deschamps C, Beauchamp G, Pagé A, Brisson J. Clinical spectrum of bronchogenic cysts of the mediastinum and lung in the adult. Ann Thorac Surg 1991; 52:6–13. REFERENCE #2: Kirmani B, Kirmani B, Sogliani F. Should asymptomatic bronchogenic cysts in adults be treated conservatively or with surgery? Interact Cardiovasc Thorac Surg. 2010 Nov; 11(5): 649-59. REFERENCE #3: Fievet L, Gossot D, de Lesquen H, Calabre C, Merrot T, Thomas P, Becmeur F, Grigoroiu M. Resection of bronchogenic cysts in symptomatic versus asymptomatic patients: an outcome analysis, The Annals of Thoracic Surgery (2020) DISCLOSURES: no disclosure on file for Gary Hochhheiser; No relevant relationships by Obieze Nwanna Nzewunwa, source=Web Response No relevant relationships by Edmund Sears, source=Web Response No relevant relationships by Hilamber Subba, source=Web Response