Ann Thorac Surg 2009;87:1919-1920. doi:10.1016/j.athoracsur.2008.10.018
© 2009 The Society of Thoracic Surgeons
Case Reports
Intrabronchial Rupture of Bronchogenic Cyst
Thiagarajamurthy Sundaramoorthi, FRCSa,*,
Ramasamy Mahadevan, MDb,
Kaliaperumal Nedumaran, MDb,
Samy Jayaraman, DTCDc,
Karthik Ramakrishnan Vaidyanathan, MSd
a Department of Cardiac and Thoracic Surgery, Billroth Hospitals, Shenoy Nagar, Chennai, India
b Department of Anesthesiology, Billroth Hospitals, Shenoy Nagar, Chennai, India
c Department of Chest Medicine, Billroth Hospitals, Shenoy Nagar, Chennai, India
d Department of Thoracic Surgery, Santosham Chest Hospital and Apollo Hospitals, Chennai, India
Accepted for publication October 10, 2008.
* Address correspondence to Dr Thiagarajamurthy, Department of Cardiac and Thoracic Surgery, Billroth Hospitals, 43 Lakshmi Talkies Rd, Shenoy Nagar, Chennai, 600030, India (Email: moorti{at}gmail.com).
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Abstract
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Bronchogenic cysts are the most common cystic masses in the mediastinum. They are generally asymptomatic and are detected incidentally on chest radiographs as a smooth homogeneous mediastinal/pulmonary opacity. Intrapleural, intraesophageal, and pericardial rupture of these cysts have been commonly reported. We report a case of life-threatening intrabronchial rupture of a subcarinal bronchogenic cyst successfully treated by an emergency thoracotomy deroofing.
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Introduction
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Bronchogenic cysts are the most common cystic masses in the mediastinum [1]. They are generally asymptomatic and are detected incidentally on chest radiographs as a smooth homogeneous mediastinal or pulmonary opacity [2]. Intrapleural, intraesophageal, and pericardial rupture of these cysts have been commonly reported [3–5]. We report a case of life-threatening intrabronchial rupture of a subcarinal bronchogenic cyst that happened during admission and was successfully treated by an emergency thoracotomy.
A 35-year-old man presented with a history of dysphagia of recent onset. Esophagoscopy was normal. Chest radiograph showed a large, smooth, homogeneous mediastinal opacity (Fig 1A). While awaiting further evaluation in the hospital, the patient had a bout of severe cough with expectoration of serous yellow material, respiratory distress, and near respiratory arrest. He was immediately intubated and ventilated. Chest radiography was repeated (Fig 1B) and showed an air fluid level in the region where there was opacity earlier. A diagnosis of ruptured mediastinal cyst was made. Computed tomography of the chest (Fig 2) showed a large subcarinal cyst with air fluid level suggestive of a rupture. Emergency thoracotomy was done through the right fifth intercostal space, and the cyst was dissected out. It was densely adherent to the right main bronchus and the esophagus. The cyst wall was opened, and its contents sucked out. A portion of the cyst wall was excised, and the adherent portion was marsupialized. On histologic examination, the cyst wall was lined with respiratory epithelium, thereby confirming it to be a bronchogenic cyst. Postoperative recovery was uneventful, and the patient was discharged home on the sixth postoperative day.
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Fig 1. (A) Chest radiograph before rupture of cyst showing well-defined, round, mediastinal shadow (arrow). (B) Chest radiograph after rupture showing air fluid level in the region where there was opacity (arrow).
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Fig 2. Computed tomography scan shows the subcarinal cyst with air inside suggestive of rupture (arrow).
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Comment
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Embryologically, bronchogenic cysts are formed when a bud separates abnormally from the developing tracheobronchial tree [1]. Early separation causes mediastinal cysts, seen commonly in the subcarinal location, and late separation causes intrapulmonary cysts. Apart from these classical sites, bronchogenic cysts may also be found in the retroperitoneum, wall of the esophagus or stomach, cardiac ventricles, or even subcutaneously [6–8]. Most of these cysts are asymptomatic. Cough and pain due to the stretch of pleura tend to occur commonly in symptomatic patients [3]. Compression on adjacent structures like esophagus, bronchus, or any of the cardiac chambers may also produce symptoms. Spontaneous rupture is a very rare complication and has often been reported to occur into the esophagus, pleural cavity, or pericardial cavity [3–5]. A literature search revealed one report of intrabronchial rupture of a bronchogenic cyst [9].
The diagnosis of a bronchogenic cyst can be suspected on plain radiographs and computed tomography [1, 2]. Definitive preoperative diagnosis by imaging alone, however, can be established only in approximately 60% of patients [2].
Diagnosis of a symptomatic mediastinal cyst mandates excision [1]. Complete excision should be done whenever possible. The presence of dense adhesions, as in our patient, may sometimes prevent complete excision, and in such cases, cauterization of the remnant mucosa has been reported to prevent recurrences [2]. Transbronchial/mediastinal aspiration has been described, but not recommended except in emergent situations or in the presence of contraindications to surgery. Aspiration can produce recurrences and malignancy from the remnant cyst wall [2].
Although management of symptomatic cysts is straightforward, the treatment of asymptomatic cysts remains debatable. Proponents of excision of even asymptomatic cysts argue that the clinical behavior of these cysts is unpredictable, and that surgical excision is the only way to confirm the diagnosis and exclude the malignancy that may rarely develop in a cyst [1]. The morbidity and mortality associated with surgery, especially with the advent of minimally invasive approaches, is minimal and is another reason stated for advocating excision [1]. We concur with this view, and based on our experience, put forth one more reason in favor of surgical excision. Our patient had sudden rupture into the airway, causing near respiratory arrest. We believe that this patient could be saved only because he was in the hospital when the rupture occurred. The potential nature of these cysts to rupture and cause life-threatening airway obstruction should be borne in mind, and any cyst close to the major airways should be considered for excision on diagnosis.
Life-threatening rupture of bronchogenic cysts into the airways can occur suddenly. This potential for rupture is a strong indication for intervention in patients diagnosed to have a mediastinal cyst close to the major airways.
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References
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- Patel SR, Meeker DP, Biscotti CV, Kirby TJ, Rice TW. Presentation and management of bronchogenic cysts in the adult Chest 1994;106:79-85.[Abstract/Free Full Text]
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- Schmidt CA, Gordon R, Ahn C. Bronchogenic cyst presenting subsequent to intrapleural rupture West J Med 1981;134:212-214.[Medline]
- Benslimane A, Bellorini M, Funck F, Guillard N, Lefevre T. Recurrent bronchogenic cyst with rupture into the pericardium. A case report. Arch Mal Coeur Vaiss 1998;91:1187-1191.[Medline]
- Khaled A, Sfia M, Fazaa B, Zermani R, Ben Jilani S, Kamoun MR. Multiple cutaneous bronchogenic cysts located on the neck and the scalp. A case report. Acta Dermatovenerol Alp Panonica Adriat 2008;17:69-71.[Medline]
- Chuang KH, Huang TW, Cheng YL, et al. Esophageal bronchogenic cyst: a rare entity Z Gastroenterol 2007;45:958-960.[Medline]
- Weinrich M, Lausberg HF, Pahl S, Schäfers HJ. A bronchogenic cyst of the right ventricular endocardium Ann Thorac Surg 2005;79:e13-e14.[Abstract/Free Full Text]
- De Nunzio MC, Evans AJ. Case report: the computed tomographic features of mediastinal bronchogenic cyst rupture into the bronchial tree Br J Radiol 1994;67:589-590.[Abstract/Free Full Text]
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Abstract
Introduction
