Intra-esophageal rupture of a bronchogenic cyst
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《血管的通路杂志》
Cardiovascular and Thoracic Surgery Department, Robert Debre Teaching Hospital, Av General Koenig, 51092 Reims, France
Abstract
Bronchogenic cysts usually are an asymptomatic disease and present as an incidental finding in the chest X-rays. They require a complete and early surgical excision to prevent complications and recurrences. We report a rare case of a voluminous symptomatic para-esophageal bronchogenic cyst complicated by an esophageal fistula. The initial video-assisted thoracoscopic surgery excision of the cyst was converted in open thoracotomy to suture the esophagus and interpose omentum. Previously, only four cases of para-esophageal bronchogenic cysts with esophageal communication have been reported.
Key Words: Cysts; Esophagus; Fistula; Bronchus
1. Case report
A 25-year-old female, with no medical past, consulted our centre for acute back pain with dysphagia for 24 h. Chest radiography revealed a significant rounded mass in the centro-posterior mediastinum. The thoracic computerized tomography (CT) diagnosed a left compressing paraesophageal bronchogenic cyst, 9x6 cm (Fig. 1). Associated gastroscopy (with no biopsy) confirmed the severe compression, showing an ulcero-necrotic esophageal wall over a 3 cm2 zone (Fig. 2). There was no clinical nor biological inflammatory syndrome. The next day she suffered from significant vomiting. Complete excision of the cyst by left video-assisted thoracoscopic surgery in emergency revealed a fistula of the bronchogenic cyst into the esophagus. There was no bronchial communication. Histological examination confirmed the benign nature of the cyst. Conversion to open posterolateral thoracotomy was decided; first, to carefully close the esophageal wall. Second, to prepare an omental flap through a retrosternal disinsertion of the diaphragm: the great omentum was pedicled at the right gastroepiploic artery, pulled up into the thoracic cavity, and sutured to cover the esophageal lesion. A nasoesophageal (just above the suture to collect oral secretions), and nasogastric aspirations were placed to protect the esophageal anastomosis. Recovery was obtained within three weeks with no sequel, except for initial food recommendations.
2. Discussion
Bronchogenic cysts are congenital anomalies of the bronchial tree, developing from the ventral part of the endodermic primitive foregut around the fourth week of gestation. In the available series, bronchogenic cysts are asymptomatic in 37 to 56%, with incidental finding in 90%, during physical or pre-employment screening [1,2]. When present, the most common symptoms are chest pain, cough, dyspnea and in the case of para-esophageal topography, dysphagia. For Patel [2], 100% of patients first asymptomatic developed complications. It possibly leads to infection, haemoptysis, haemorrhage within the cyst, superior veina cava obstruction, pneumothorax, pleural effusion, carcinomatous change, arrhythmias [3]. Pain is caused by irritation of the pleura more than by compression. Complications are more dependent on the location of the cyst than on its volume [4].
Preoperative diagnosis is considered by chest computerized tomography (CT) or magnetic resonance imaging (MRI), showing the cystic nature of the mediastinal mass [5]. But if the bronchogenic cyst is infected or high in protein or calcium content, differential diagnosis with solid mass is uncertain: metastatic tumor, lymphoma, teratoma, embryonal sarcoma, lymphadenopathy, pulmonary sequestrations, hemangiomata and lipoma. And, with parenchymal cysts, the differential diagnosis includes neoplasm, granuloma, vascular malformation, lung abscess, infected bullae, and hydatid cyst [2]. Thus, in case of difficult preoperative diagnosis of a para-esophageal mass, endoscopic ultrasound is a safe and very useful procedure to clearly distinguish cystic from solid masses as well as defining the intra-extramural extent of the lesion [6]. Gastroscopies and bronchoscopies exclude a communication of the cyst with the esophagus or the tracheobronchial tree, but no biopsy specimen should be taken so as not to complicate following surgical excision [1].
Surgical indication is clear today. As in our case, resection of symptomatic cysts may be associated with greater operative difficulty: all suspected bronchogenic cysts must be removed in operable candidates [2]. And moreover, a complete surgical excision is the only way to get a perfect histological diagnosis. It also prevents from complications and recurrences [4,7].
The surgical procedure [1–4] is a three trocars technique video-assisted thoracoscopy, using the fifth intercostal space (anterior and posterior axillary, and midclavicular lines), by right or left lateral position after double-lumen intubation. Esophageal mucosal integrity should be checked intraoperatively by air insufflation through the nasogastric tube or permanent transillumination through the esophagoscope. Complete exploration of the cavity precedes resection. During the dissection, vagal, phrenic and recurrent laryngeal nerves must be preserved. Complete resection is the best treatment; but if the cyst is adherent to vital structures, a small patch of cystic wall is left in place, after destruction of the mucosal lining by electocautery to avoid recurrences [8]. The need for a posterolateral thoracotomy is possible in different situations: vascular injuries, extensive pleural adhesions, central topography of the cyst, esophageal [10] or tracheobronchial fistula. When such a communication is discovered, firstly resection of the inflammatory tissue is done; intrapulmonary lesions require segmental or lobar resection. The esophagus is closed by a 4-0 interrupted absorbable suture in two levels, transversally if possible to avoid stenosis. Following completion of repair, a viable tissue such as a strip of thickened pleura, a pericardial fat, a pedicled intercostal muscle flap or an omentum pedicle should be interposed to buttress the suture line [9]. We think that post-operative management as described in our case is very important to protect the esophageal anastomotic region and avoid recurrence of the fistula. Alternative techniques to complete surgical excision are not recommended anymore, except as temporary measures in case of acute compression or inoperable symptomatic patients: percutaneous, transtracheobronchial fine needle aspiration (FNA) or mediastinoscopic puncture [4,7].
References
Cioffi U, Bonavina L, De Simone M, Santambrogio L, Pavoni G, Testori A, Peracchia A. Presentation and surgical management of bronchogenic and esophageal duplication cysts in adults. Chest 1998;113:1492–1496.
Patel SR, Meeker DP, Biscotti CV, Kirby TJ, Rice TW. Presentation and management of bronchogenic cysts in the adult. Chest 1994;106:79–85.
Weber T, Roth TC, Beshay M, Herrmann P, Stein R, Schmid RA. Video-assisted thoracoscopic surgery of mediastinal bronchogenic cysts in adults: a single-center experience. Ann Thorac Surg 2004;78:987–991.
Ribet ME, Copin MC, Gosselin B. Bronchogenic cysts of the mediastinum. J Thorac Cardiovasc Surg 1995;109:1003–1010.
Zambudio AR, Lanzas JT, Calvo MJR, Fernández PJ, Paricio PP. Non-neoplastic mediastinal cysts. Eur J Cardiothorac Surg 2002;22:712–716.
Lim LL, Ho KY, Goh PM. Preoperative diagnosis of a paraesophageal bronchogenic cyst using endosonography. Ann Thorac Surg 2002;73:633–635.
Hasegawa T, Murayama F, Endo S, Sohara Y. Recurrent bronchogenic cyst 15 years after incomplete excision. Interactive Cardiovasc Thorac Surg 2003;2:685–687.
Martinod E, Pons F, Azorin J, Mouroux J, Dahan M, Faillon JM, Dujon A, Lajos PS, Riquet M, Jancovici R. Thoracoscopic excision of mediastinal bronchogenic cyst: results in 20 cases. Ann Thorac Surg 2000;69:1525–1528.
Mangi AA, Gaissert HA, Wright CD, Allan JS, Wain JC, Grillo HC, Mathiesen DJ. Benign bronchoesophageal fistula in the adult. Ann Thorac Surg 2002;73:911–915.
Sasaki K, Tanaka S, Koizumi K, Shioda M, Fukushima T, Shoji T, Iwakiri K, Nomura T, Kawamoto M, Tamura K. A case of paraesophageal bronchogenic cyst with esophageal communication. Kyobu Geka 1992;45:813–816.(Olivier N. Pages, Sylvain)
Abstract
Bronchogenic cysts usually are an asymptomatic disease and present as an incidental finding in the chest X-rays. They require a complete and early surgical excision to prevent complications and recurrences. We report a rare case of a voluminous symptomatic para-esophageal bronchogenic cyst complicated by an esophageal fistula. The initial video-assisted thoracoscopic surgery excision of the cyst was converted in open thoracotomy to suture the esophagus and interpose omentum. Previously, only four cases of para-esophageal bronchogenic cysts with esophageal communication have been reported.
Key Words: Cysts; Esophagus; Fistula; Bronchus
1. Case report
A 25-year-old female, with no medical past, consulted our centre for acute back pain with dysphagia for 24 h. Chest radiography revealed a significant rounded mass in the centro-posterior mediastinum. The thoracic computerized tomography (CT) diagnosed a left compressing paraesophageal bronchogenic cyst, 9x6 cm (Fig. 1). Associated gastroscopy (with no biopsy) confirmed the severe compression, showing an ulcero-necrotic esophageal wall over a 3 cm2 zone (Fig. 2). There was no clinical nor biological inflammatory syndrome. The next day she suffered from significant vomiting. Complete excision of the cyst by left video-assisted thoracoscopic surgery in emergency revealed a fistula of the bronchogenic cyst into the esophagus. There was no bronchial communication. Histological examination confirmed the benign nature of the cyst. Conversion to open posterolateral thoracotomy was decided; first, to carefully close the esophageal wall. Second, to prepare an omental flap through a retrosternal disinsertion of the diaphragm: the great omentum was pedicled at the right gastroepiploic artery, pulled up into the thoracic cavity, and sutured to cover the esophageal lesion. A nasoesophageal (just above the suture to collect oral secretions), and nasogastric aspirations were placed to protect the esophageal anastomosis. Recovery was obtained within three weeks with no sequel, except for initial food recommendations.
2. Discussion
Bronchogenic cysts are congenital anomalies of the bronchial tree, developing from the ventral part of the endodermic primitive foregut around the fourth week of gestation. In the available series, bronchogenic cysts are asymptomatic in 37 to 56%, with incidental finding in 90%, during physical or pre-employment screening [1,2]. When present, the most common symptoms are chest pain, cough, dyspnea and in the case of para-esophageal topography, dysphagia. For Patel [2], 100% of patients first asymptomatic developed complications. It possibly leads to infection, haemoptysis, haemorrhage within the cyst, superior veina cava obstruction, pneumothorax, pleural effusion, carcinomatous change, arrhythmias [3]. Pain is caused by irritation of the pleura more than by compression. Complications are more dependent on the location of the cyst than on its volume [4].
Preoperative diagnosis is considered by chest computerized tomography (CT) or magnetic resonance imaging (MRI), showing the cystic nature of the mediastinal mass [5]. But if the bronchogenic cyst is infected or high in protein or calcium content, differential diagnosis with solid mass is uncertain: metastatic tumor, lymphoma, teratoma, embryonal sarcoma, lymphadenopathy, pulmonary sequestrations, hemangiomata and lipoma. And, with parenchymal cysts, the differential diagnosis includes neoplasm, granuloma, vascular malformation, lung abscess, infected bullae, and hydatid cyst [2]. Thus, in case of difficult preoperative diagnosis of a para-esophageal mass, endoscopic ultrasound is a safe and very useful procedure to clearly distinguish cystic from solid masses as well as defining the intra-extramural extent of the lesion [6]. Gastroscopies and bronchoscopies exclude a communication of the cyst with the esophagus or the tracheobronchial tree, but no biopsy specimen should be taken so as not to complicate following surgical excision [1].
Surgical indication is clear today. As in our case, resection of symptomatic cysts may be associated with greater operative difficulty: all suspected bronchogenic cysts must be removed in operable candidates [2]. And moreover, a complete surgical excision is the only way to get a perfect histological diagnosis. It also prevents from complications and recurrences [4,7].
The surgical procedure [1–4] is a three trocars technique video-assisted thoracoscopy, using the fifth intercostal space (anterior and posterior axillary, and midclavicular lines), by right or left lateral position after double-lumen intubation. Esophageal mucosal integrity should be checked intraoperatively by air insufflation through the nasogastric tube or permanent transillumination through the esophagoscope. Complete exploration of the cavity precedes resection. During the dissection, vagal, phrenic and recurrent laryngeal nerves must be preserved. Complete resection is the best treatment; but if the cyst is adherent to vital structures, a small patch of cystic wall is left in place, after destruction of the mucosal lining by electocautery to avoid recurrences [8]. The need for a posterolateral thoracotomy is possible in different situations: vascular injuries, extensive pleural adhesions, central topography of the cyst, esophageal [10] or tracheobronchial fistula. When such a communication is discovered, firstly resection of the inflammatory tissue is done; intrapulmonary lesions require segmental or lobar resection. The esophagus is closed by a 4-0 interrupted absorbable suture in two levels, transversally if possible to avoid stenosis. Following completion of repair, a viable tissue such as a strip of thickened pleura, a pericardial fat, a pedicled intercostal muscle flap or an omentum pedicle should be interposed to buttress the suture line [9]. We think that post-operative management as described in our case is very important to protect the esophageal anastomotic region and avoid recurrence of the fistula. Alternative techniques to complete surgical excision are not recommended anymore, except as temporary measures in case of acute compression or inoperable symptomatic patients: percutaneous, transtracheobronchial fine needle aspiration (FNA) or mediastinoscopic puncture [4,7].
References
Cioffi U, Bonavina L, De Simone M, Santambrogio L, Pavoni G, Testori A, Peracchia A. Presentation and surgical management of bronchogenic and esophageal duplication cysts in adults. Chest 1998;113:1492–1496.
Patel SR, Meeker DP, Biscotti CV, Kirby TJ, Rice TW. Presentation and management of bronchogenic cysts in the adult. Chest 1994;106:79–85.
Weber T, Roth TC, Beshay M, Herrmann P, Stein R, Schmid RA. Video-assisted thoracoscopic surgery of mediastinal bronchogenic cysts in adults: a single-center experience. Ann Thorac Surg 2004;78:987–991.
Ribet ME, Copin MC, Gosselin B. Bronchogenic cysts of the mediastinum. J Thorac Cardiovasc Surg 1995;109:1003–1010.
Zambudio AR, Lanzas JT, Calvo MJR, Fernández PJ, Paricio PP. Non-neoplastic mediastinal cysts. Eur J Cardiothorac Surg 2002;22:712–716.
Lim LL, Ho KY, Goh PM. Preoperative diagnosis of a paraesophageal bronchogenic cyst using endosonography. Ann Thorac Surg 2002;73:633–635.
Hasegawa T, Murayama F, Endo S, Sohara Y. Recurrent bronchogenic cyst 15 years after incomplete excision. Interactive Cardiovasc Thorac Surg 2003;2:685–687.
Martinod E, Pons F, Azorin J, Mouroux J, Dahan M, Faillon JM, Dujon A, Lajos PS, Riquet M, Jancovici R. Thoracoscopic excision of mediastinal bronchogenic cyst: results in 20 cases. Ann Thorac Surg 2000;69:1525–1528.
Mangi AA, Gaissert HA, Wright CD, Allan JS, Wain JC, Grillo HC, Mathiesen DJ. Benign bronchoesophageal fistula in the adult. Ann Thorac Surg 2002;73:911–915.
Sasaki K, Tanaka S, Koizumi K, Shioda M, Fukushima T, Shoji T, Iwakiri K, Nomura T, Kawamoto M, Tamura K. A case of paraesophageal bronchogenic cyst with esophageal communication. Kyobu Geka 1992;45:813–816.(Olivier N. Pages, Sylvain)