A case of bronchoesophageal fistula diagnosed 32 years after surgery for pulmonary sequestration

• Published in: Nihon Kokyuki Geka Gakkai zasshi (Kyoto, 1992) Vol. 38; no. 5; pp. 426 - 432
• Main Authors: Makita, Ayaka, Seki, Yukio, Ichihara, Tomofumi, Yamada, Katsuo, Yasuda, Ayuko
• Format: Journal Article
• Language: Japanese; English
• Published: The Japanese Association for Chest Surgery 15.07.2024
• Subjects: Braimbridge type I; Braimbridge type IV; congenital bronchoesophageal fistula; surgery
• ISSN: 0919-0945; 1881-4158
• DOI: 10.2995/jacsurg.38.426

The patient was a woman in her 40s with a history of drainage surgery for pulmonary pyogenic disease at the age of 14 years, and a right lower lobectomy for pulmonary sequestration at 17 years of age. Since then, she had been asymptomatic; however, at the time of a comprehensive health check-up, an upper gastrointestinal tract radiograph revealed extravasation of the contrast agent into the bronchial tubes, prompting a referral for further evaluation. During esophagoscopy, an esophageal diverticulum was observed in the middle esophagus, 25 cm from the upper incisor. Subsequent gastrografin contrast imaging from the diverticulum revealed leakage of the contrast agent into the bronchus. On bronchoscopy, a needle-sized fistula was identified approximately 1 cm proximal to the resection margin of the right lower lobe. Based on these findings, a diagnosis of bronchoesophageal fistula was made. Surgical intervention was performed through video-assisted thoracoscopic surgery (VATS) via an open approach with division of the fistula using an automated suture machine, followed by reinforcement of the divided site with a pedicled intercostal muscle flap. During the surgery, minimal inflammation was observed in the vicinity of the fistula. Therefore, although histological analysis of the fistula site was not performed in this case, a congenital tracheoesophageal fistula could not be conclusively ruled out. The fistula site was located in the middle trunk, close to the right B6 resection margin. As the sequestrated lung had been resected, its relationship with the resected lung segments remains uncertain. If congenital, this is an exceedingly rare case, considering the presence of the esophageal diverticula and course of the disease in the patient. This case brings to our attention the importance of considering the rare but possible occurrence of bronchoesophageal fistula when planning treatment for pulmonary sequestration.