The case of the burping man
Gordon K. Mak
A 73-year-old man presented to his primary care provider complaining of belching, dysphagia, and productive cough of recent onset. His medical history included stage IV lung cancer first noted 1 year ago on a chest radiograph obtained during a workup for cough. At that time, a chest CT scan revealed a 4 x 4-cm cavitary mass in the right upper lobe with extension into the paratracheal region and marked esophageal wall thickening. A pathologic diagnosis of squamous cell carcinoma was made based on mediastinal biopsy of the right paratracheal lymph nodes. Other findings detected with mediastinoscopy were a 3-cm soft tissue mass along the posterior pleura of the left lower lobe with adjacent destruction of the left ninth rib, multiple enlarged lymph nodes throughout the mediastinum, a 1.5 cm nodular mass in the posterior segment of the left lower lobe, all of which may suggest stage IV bronchogenic carcinoma.
The oncologist treated the patient for presumed bronchogenic carcinoma. He received 6 cycles of chemotherapy with paclitaxel and carboplatin. The lesions decreased with chemotherapy.
The patient also had a history of alcohol abuse, chronic obstructive pulmonary disease, and smoking, but he had quit smoking 1 year previously. He denied recurrent respiratory infections. Physical examination and laboratory evaluation were unremarkable.
A repeated chest CT scan indicated persistent involvement of the paratracheal region with marked esophageal wall thickening (Figure 1). The right upper lobe opacity had resolved. Upper GI endoscopy revealed circumferential narrowing of the esophagus by an extrinsic mass with overlying ulceration. Findings on the examination distal to the mass were normal. Bronchoscopy was then performed(Figure 2).
How would you interpret these findings?
What do you think the diagnosis will be?
Making the diagnosis
The chest CT scan suggested a tracheoesophageal fistula. Fiberoptic bronchoscopy revealed extrinsic compression of the posterior tracheal wall due to a large mass. The mucosa appeared hypervascular with submucosal infiltration. A small tracheoesophageal fistula was visualized on the posterior wall approximately 5 cm above the carina, with air and secretions oozing through this defect.
All segmental bronchi visualized in the right and left lungs were of normal appearance. Unfortunately, methylene blue was not available during the bronchoscopy to more precisely determine the diagnosis. At that time, a contrast esophagram was not performed for palliative reasons because the patient had advanced cancer with pro found dysphagia.
Tracheoesophageal fistulae are severe lesions that lead to serious and often fatal pulmonary complications. These lesions are predominantly iatrogenic, occurring in the course of tracheal intubation for resuscitation, or of malignant origin with invasion of esophageal and tracheal walls. 
Tracheoesophageal fistulae are a serious complication of cancer. They usually develop in the esophagus, lungs, or tracheobronchial tree. Repetitive aspiration and pneumonia can lead to rapid deterioration and death. The prognosis is dismal.
Tracheobronchial injuries may be caused by external injuries from blunt or penetrating trauma or by internal injuries subsequent to in halation of fumes or gases or aspiration of liquids or foreign bodies. These injuries are rare but can be life-threatening. Acquired, nonmalignant tracheoesophageal fistulae usually result from erosion of the tracheal and esophageal walls by endotracheal or tracheostomy tube cuffs, especially when a rigid nasogastric tube is in place. This life threatening condition is infrequent with the use of high-volume and low-pressure cuffs.
The clinical features of tracheoesophageal fistulae are often non specific. The patient may complain of increased sputum production, gas, and frequent respiratory infections. Imaging studies play a pivotal role in timely diagnosis. [2-4] Contrast esophagrams with non ionic contrast typically detect an esophageal perforation if the symptoms or history suggests the diagnosis.
CT scanning should be per formed early in the workup. It is essential to recognize the CT features of esophageal perforation, which may include the presence of extraluminal air, periesophageal fluid, esophageal thickening, and extraluminal contrast. A communication may be noted between the esophagus and the trachea. These findings may be the first clue to the correct diagnosis. 
Bronchoscopy is useful. It may reveal the fistula or the passage of secretions or air. Oral administration of methylene blue has also been used as a diagnostic aid during bronchoscopy. 
Difficulty in treatment arises from the need to manage both the consequences of communication between the trachea and esophagus and the aftermath of the illness responsible for fistula.  For nonmalignant tracheoesophageal fistulae, surgical correction, including esophageal closure, segmental tracheal resection, and primary reconstruction, is the preferred option.  For malignant fistulae, the mainstay of treatment is palliative and is directed at stopping soilage of the respiratory tract.
The type of therapy is dictated by the performance status of the patient at presentation. Options include the division and closure of the fistula and esophageal exclusion followed by gastric or colic bypass or intubation.  Esophageal bypass offers the best palliation for those able to tolerate the procedure.  Surgical bypass of the lesion in malignant tracheoesophageal fistulae is associated with 25% to 61% mortality. 
Other treatments include enterostomies, esophageal endoprostheses, and supportive care. Expansile metallic esophageal stent placement is particularly useful in patients who have fistulae caused by malignancy and who are not responsive to dilation.  The reported mortality rates from these palliative procedures are almost identical. Since esophageal endoprostheses can be placed with fewer major complications, this procedure gained popularity in the past decade, making it the preferred option. Surgical correction can be considered on a case-by-case basis. 
Outcome in this case
The patient described here complained of increased belching, which is likely to have been secondary to aspiration of secretions and passage of air through the tracheoesophageal fistula. The diagnostic feature was very subtle on CT scans. Bronchoscopy visualized the fistula and demonstrated the passage of secretions and air. Esophageal biopsy confirmed well-differentiated squamous cell carcinoma, consistent with metastasis from the lung and with the pathology seen in previous biopsy of the right paratracheal lymph node. An esophageal stent was eventually placed. A contrast esophagram was then performed, which showed a well-seated stent and no evidence of fistula. The patient continued to have dysphagia, and a gastrostomy tube was inserted. His health continued to decline, and he subsequently died.
The authors are affiliated with the division of pulmonary and critical care medicine at Stanford University School of Medicine and the Veterans; Affairs Palo Alto Health Care System, in Palo Alto, California. Dr Mak is a fellow, Dr Sarinas is assistant professor of medicine, Dr Kuschner is assistant professor of medicine, and Dr Chitkara is associate professor of medicine.
Dr Louie is professor of medicine in the division of pulmonary and critical care medicine at the University of California, Davis, School of Medicine in Sacramento. Dr Lillington is professor of medicine emeritus at the same institution and clinical professor of medicine at Stanford University School of Medicine in California; he is a member of the editorial board of The Journal of Respiratory Diseases.
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