Discussion
We experienced a relatively rare case of recurrent TEF that was found 10 years after the initial repair. We herein discuss the reason why the recurrent TEF had not been identified and the surgical technique that we used to repair the patient’s recurrent TEF.
The patient had subglottic stenosis, which made it difficult to observe the trachea. In addition to the subglottic stenosis, both bronchial asthma and recurrent hiatus hernia were thought to have caused the patient’s respiratory symptoms due to frequent pneumonia. These factors may have caused the patient’s recurrent TEF to remain unidentified. Recurrence was first suspected when a small volume of nutritional supplement that had been administered via gastrostomy was aspirated from the tracheostomy tube after the laryngotracheal separation. In general, although many cases of recurrent TEF are diagnosed by a contrast study, some require further examination by endoscopy with or without a dye test [5, 6, 8, 11]. In the present case, bronchoscopy (which is safer and precise) was not possible prior to tracheostomy due to the patient’s subglottic stenosis. As shown in Fig. 2, esophagoscopy and bronchoscopy combined with a dye test enabled us to diagnose the recurrent TEF.
In general, almost all patients with recurrent TEF have episodes of leakage and/or tracheal injuries [4–6, 8]. The recurrent fistula usually opens in the trachea at the site of the original fistula site or tracheal injury [4–6]. In this case, there was no leakage or tracheal injury at the site of the primary repair. In contrast with other cases, the site of recurrence was found to be high in the cervical area. Thus, we suspected that the patient had both proximal and distal tracheoesophageal fistula (gross type D), and the upper pouch fistula was missed in the primary operation [12, 13]. However, as shown in Fig. 2, a pit was identified just beside the anastomotic line. The anastomotic line in the esophagus had shifted to the cervical area because of the recurrent hiatus hernia. We therefore judged that the original fistula (proximal tracheoesophageal fistula of gross type D) in the upper pouch was not found at this time. We also suspected that the trachea had been injured during laryngotracheal separation. However, during the repair of the recurrent TEF, the fistula was found to be located in an area that had not been touched during the laryngotracheal separation. Based on the above-noted findings, we diagnosed the patient with recurrent TEF.
In the present case, the TEF might have recurred at the age of 9, when the patient started suffering from frequent and severe pneumonia. The recurrent hiatus hernia caused GER and the poor clearance of refluxed gastric juice in the esophagus. Prolonged acid contact causing inflammation and erosion around the anastomotic site is thought to have been important in the pathogenesis of TEF in the present case.
There are several options for repairing recurrent TEF. Endoscopic techniques using adhesives have been presented [14–16]. This method is reported to be effective when the fistula tract is first de-epithelialized using several types of devices and then fibrin glue or a similar agent is injected into the tract of the fistula [5, 14–16]. However, it is thought that this technique would have been difficult in the present case because the tract was not detected clearly and because it was not possible to place a catheter under endoscopy. We therefore opted for open surgical closure. The placement of viable tissue between the suture lines may help to prevent the further recurrence of TEF [5, 6, 9, 10]. Previous studies have described the use of the pericardium, pleura, intercostal flap, cervical muscle flap, cartilage, and lymph nodes for this purpose [5, 6, 9, 10]. The flap can be harvested from the same surgical site with its own blood supply. In the present case, a sternothyroid muscle flap was chosen because the recurrent fistula was in the cervical area. In fact, in the present case, the muscle flap was easily and safely interposed between the divided ends of the fistula with a good blood supply. Based on our experience, the interposition of a muscle flap using the sternothyroid muscle is thought to be a good option for reoperation to repair a recurrent TEF in the cervical area.