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J Thorac Cardiovasc Surg 2006;131:916-917
© 2006 The American Association for Thoracic Surgery
Brief Communication |
a Servei de Cirurgia General, Abdominal, i Toràcica, Hospital del Mar, Institut Municipal d'Assitència Sanitària, Barcelona, Spain.
b Unitat d'Endoscòpia Digestiva, Hospital del Mar, Institut Municipal d'Assitència Sanitària, Barcelona, Spain.
c Unitat de Diagnòstic per l'Imatge, Hospital del Mar, Institut Municipal d'Assitència Sanitària, Barcelona, Spain.
Received for publication September 30, 2005; accepted for publication October 26, 2005. * Address for reprints: Rafael Aguiló, MD, PhD, Servei de Cirurgia General, Abdominal, i Toràcica, Hospital del Mar, Institut Municipal d'Assitència Sanitaria, Barcelona, Spain. (Email: 95183{at}imas.imim.es).
Tracheoesophageal or bronchoesophageal fistula (T/BEF), the abnormal communication between the esophagus and the tracheobronchial tree, may present at any time during the life span. In the neonate, T/BEF is a congenital malformation associated with some form of esophageal atresia in the vast majority of cases.
1,2
Later in life, most T/BEFs are acquired.
3,4
However rare, if the congenital T/BEF is not associated with esophageal atresia, it may remain silent until it eventually becomes symptomatic. Herein, we report the clinical case of a 53-year-old woman whose congenital T/BEF was diagnosed and treated at our institution.
Clinical Summary
The patient was referred to us as a result of a self-limited episode of minor hemoptysis of sudden onset. At anamnesis, she explained that she had experienced a pain in the upper abdomen 2 years before. Gallbladder stone disease and unilateral renal agenesia were diagnosed by echography, and chronic gastritis was diagnosed by esophagogastroscopy. She was then cholecystectomized without postoperative complications. She did not disclose any other previous disease or complaint. Physical examination was normal on admission. Red blood cell, white blood cell, and platelet counts, coagulation profile; routinary serum; and urine analysis results were all within the normal range. Chest radiography and a computed tomographic scan of the thorax displayed an irregular area of lung parenchyma condensation in the upper segment of the left lower lobe. On fiberoptic bronchoscopy, the tracheobronchial tree was normal except for the presence of a blood clot that partly occupied the lumen of the left lower lobe bronchus. Bronchial washing specimens were sent for cytologic analysis, Gram and acid-fast stain, and bacterial and mycobacterial culture; all yielded negative results. Esophagogastroduodenoscopy revealed the presence of a double hole in the left wall of the middle esophagus. Bouts of cough ensued every time the holes were instilled with serum saline, thus establishing the diagnosis of T/BEF. Initial treatment consisted of full oral fasting and total enteral nutrition for 3 weeks. Then, an attempt was made to close the fistula by endoscopic instillation of fibrin glue. However, an esophagogram showed the persistence of the T/BEF (Figure 1), and the patient was scheduled for an open surgical procedure. Dissection of the fistula, closure of both ends with an automatic stapler, division of the tract, handmade reinforcement of both sutures, and interposition of a pedicled pleural flap were performed through a left posterolateral thoracotomy. Postoperative evolution was normal. A second esophagogram showed no spilling of the contrast. The patient restarted oral feeding on the sixth postoperative day.
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Congenital T/BEF seldom presents in adulthood because it is rarely left undiagnosed in the neonate: it is often associated with some other bronchopulmonary and/or foregut malformation (mainly with esophageal atresia).
1,2
It is only in the absence of this association that a congenital T/BEF may remain silent and not be diagnosed until much later, when it becomes symptomatic. There is no explanation for the possible mechanism(s) that make a T/BEF remain silent for long. Occlusion by an imperforated web and/or the ascending (antigravitational) direction of the tract have been advocated.
1,2
Besides, it is not known what the precipitating events are that cause symptoms to start. When this happens, symptoms are most often respiratory, acute, or chronic. Bouts of cough when swallowing, chronic wheezing, recurrent bronchopulmonary infections, hemoptysis, and hematemesis, either isolated or combined, should raise the suspicion of T/BEF. Diagnosis is made by endoscopy, esophagography, or both. Computed tomographic scanning of the chest is useful because repeated lung infections can lead to bronchiectasis. Fiberoptic bronchoscopy is also mandatory. The congenital nature of a T/BEF is admitted when an acquired cause is reasonably excluded. At pathology examination, an epithelium-lined tract with muscularis mucosae is conclusive. Once diagnosis is made, treatment should be aggressive and promptly instituted, because the acute worsening of the patient's condition due to potentially life-threatening complications, such as massive hemoptysis, is possible at any time. Treatment is as described previously. Endoscopic occlusion is an option to try on an individual basis. If bronchiectasis is present, then resection of the affected segment(s) or lobe(s) should be performed.
3,4
The case we present corresponded to a type I congenital T/BEF in Braimbridge and Keith's classification.
5
It consists of a tract connecting the esophagus and a lobar or segmental bronchus, associated with a congenital esophageal diverticulum. Our patient also had unilateral renal agenesia. We could not identify any precipitating event. Notably, the patient had undergone an esophagogastroscopy 2 years before that did not disclose esophageal disease.
References
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