Volume 3, Issue 4 , Pages 161-164, December 2008
Acquired tracheo-esophageal fistula in a child caused by an unsuspected esophageal foreign body
Article Outline
Summary
Acquired tracheo-esophageal fistula (TEF) is very infrequent in the pediatric age group and is usually caused by esophageal foreign body impaction. Bronchoscopy plays an essential role in the diagnostic work-up of this rare complication. Delayed surgical closure with muscle flap interposition is the treatment of choice if spontaneous healing does not occur.
The authors present the case of a 5-year-old boy with an acquired TEF caused by a very unusual esophageal foreign body, treated surgically. Management of this rare entity is discussed.
Keywords: Tracheo-esophageal fistula, Children, Bronchoscopy, Esophageal foreign body
1. Introduction
Acquired tracheo-esophageal fistula (TEF) is an infrequent disease described mainly in adults. Very sick aged patients with long standing cuffed tracheotomies, in whom a large nasogastric tube is in place, are the usual candidates to develop this severe complication. Other etiologies such as: burn injuries, trauma, infection, and malignancy have also been described [1], [2], [3]. In children, acquired TEF is extremely rare and it is usually caused by esophageal foreign body impaction, being disc batteries responsible for most of the reported cases [4], [5], [6], [7].
Herein, we present the case of a 5-year-old boy with an acquired TEF caused by a very unusual esophageal foreign body. Diagnostic tools and management patterns are discussed.
2. Case report
A 5-year-old boy was admitted to a local hospital with a 3-week history of persistent cough and odynophagia. A chest X-ray showed bilateral basal infiltrates and Streptococcus pneumoniae was isolated in blood cultures, so antibiotherapy was started. His general condition improved in the following days and he was discharged 2 weeks after admission although cough had not disappeared completely, being more significant when eating. For this reason he was scheduled for a gastro-esophageal contrast study on an outpatient basis. The barium swallow showed a tracheo-esophageal fistula next to the thoracic inlet, and the patient was immediately transferred to our institution.
Bronchoscopy showed a white foreign body 4.5
cm distal to the vocal cords partially obstructing the tracheal lumen. Rigid bronchoscopic removal was attempted but proved unsuccessful, so an esophagoscopy was then performed. A hard white plastic cone-type foreign body (tooth-paste tube cap) was found on the anterior esophagus 15cm distal to the incisors. It was removed with an appropriate flexible endoscopic forceps disclosing a large tracheo-esophageal fistula (1.5cm diameter) with mucosal edema and granulation tissue (Fig. 1a and b). A subsequent CT scan of the neck and chest confirmed the endoscopic findings (Fig. 2).
Fig. 1.
(a) Endoscopic removal of a cone type plastic foreign body in the esophagus. (b) Bronchoscopic view of the tracheo-esophageal fistula (arrow).
Fig. 2.
Chest CT: the large communication between the trachea and the esophagus can be observed.
Surgical treatment was deferred due to the inflammatory changes found in both the tracheal and esophageal mucosas at the TEF site. A percutaneous endoscopic gastrostomy was performed for feeding purposes and antireflux medical treatment was instituted. Six weeks later, mucosal inflammation and granulation tissue had completely disappeared and the fistula's size remained the same. A left cervicothomy provided excellent exposure of the tracheo-esophageal fistula which was circumferentially dissected and divided. Both the esophageal and tracheal openings were closed with interrupted absorbable sutures and a cervical muscle flap (sternohyoid) was interposed between them. The patient was extubated in the operating room and the postoperative course was uneventful. An esophagogram on the 7th postoperative day showed no evidence of fistula or stenosis, so oral feeding was resumed and the gastrostomy tube was removed soon after. Bronchoscopy performed 1 month postdischarge showed a normal trachea. The patient is asymptomatic and doing well 3 years after the surgical correction.
3. Discussion
Foreign body ingestion is a frequent hazard in the early years of life, and when esophageal impaction occurs prompt endoscopic removal should be performed. Coins and disc batteries have been claimed to be the most common foreign bodies removed at esophagoscopy [4], [8], [9]. Morbidity is usually low, although button batteries are more prone to cause complications if they get lodged in the esophagus [4], [5], [9]. The incidence of esophageal perforation has been estimated to be around 1% [10]. Miller et al. [8] reported 18 patients with esophageal perforation out of 522 with esophageal foreign bodies (3%), although only one patient showed a “classic” or complete perforation. No patient in their 12 year retrospective review presented with TEF.
Tracheo-esophageal fistula due to esophageal foreign body impaction is an extremely rare complication, caused by disc batteries in the majority of the reported cases [4], [5], [6], [7]. Perforation can occur in a few hours due to the special characteristics of these chemical devices [5]. In our patient, perforation and fistula formation took several days because of the progressive pressure exercised in the esophageal wall by the plastic cone type foreign body resulting in full-thickness necrosis.
Diagnosis is challenging because children may present with a wide variety of symptoms, both respiratory and digestive. Laringo-tracheo-bronchitis, asthma, or respiratory infection are the usual initial diagnosis when presenting to the primary care physician or to the emergency room [8]. Diagnosis is even more difficult in those cases in which the foreign body is not radiopaque and ingestion is unwitnessed, as happened in our patient. An esophagogram disclosed the unexpected TEF but the etiology could not be identified until endoscopic explorations (airway and digestive) were performed. In our experience, bronchoscopy has been very useful in evaluating the size and situation of the fistula, and the degree of mucosal inflammation. Direct transillumination when performing tracheo-bronchoscopy with neck hyperextension permits precise location, above or below the thoracic inlet, in order to plan the appropriate surgical approach. Imamoglu et al. [4] advocate the use of three-dimensional multislice CT and virtual bronchoscopy instead of standard bronchoscopy. Although the anatomic features of the TEF may be accurately described by this technique, virtual bronchoscopy cannot evaluate the degree of mucosal inflammation nor identify the presence of granulation tissue. In experienced hands, bronchoscopy carries very low risks and, in our opinion, should be performed in any suspected case of acquired TEF.
Some authors favour conservative management for acquired TEF [6], [7], [11]. Spontaneous closure may occur in patients with small fistulas and no underlying diseases, taking as long as 4–11 weeks. Large TEF, 15mm diameter or more, are less prone to spontaneous healing and should be managed surgically. Nevertheless, most authors favour a delayed surgical closure because of concern with active mucosal inflammation and granulation tissue at the TEF site [4], [5], [12]. During this waiting time period the patient is fed by means of a gastrostomy, or a jejunostomy tube, and bronchoscopy can be performed to evaluate inflammatory changes and eventual spontaneous closure. Simple division of the fistula and interposition of a muscle flap between the trachea and the esophagus is the treatment of choice if spontaneous healing does not occur [4], [12]. Cervical esophagostomy and gastrostomy with delayed colon interposition is indicated in cases of recurrent TEF [5].
In conclusion, persisting respiratory symptoms and/or feeding problems after an adequately treated respiratory infection should rise the suspicion of an unsuspected chronic esophageal or tracheal foreign body, and prompt endoscopic exploration should be carried. If a TEF is diagnosed, conservative management may be initially tried but if spontaneous closure does not occur operative treatment is indicated.
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PII: S1871-4048(08)00022-1
doi:10.1016/j.pedex.2008.02.004
© 2008 Elsevier Ireland Ltd. All rights reserved.
Volume 3, Issue 4 , Pages 161-164, December 2008
