Tracheoesophageal Fistula as a Complication of Prolonged Ventilation in COVID-19: Description of Reconstruction and Review of the Literature

Abstract

Infection with COVID-19 pneumonia may necessitate intubation and mechanical ventilation. Viral inflammation and pressure necrosis may lead to scarring, stenosis, and in severe cases, fistula formation. Nonmalignant tracheoesophageal fistulas (TEF) represent a surgical challenge and may necessitate locoregional tissue transfer and tracheal resection to prevent recurrence and maintain airway patency. We present a case of TEF in a 63-year-old female secondary to prolonged mechanical ventilation in the setting of COVID pneumonia, detailing the clinical findings and surgical repair. Primary closure of the esophageal defect with pectoralis major muscle flap onlay and tracheal resection, with median sternotomy for access, provided successful intervention, allowing for subsequent tracheostomy decannulation and return to a complete oral diet. This case offers further evidence of the increased risk of airway complications in COVID-19 infection and provides otolaryngologists with an example of a rare surgical approach useful in management.

Introduction

The high prevalence of laryngotracheal injury following COVID-19 infection has made postinfectious sequalae commonplace in the otolaryngology clinic. Diagnosis is often delayed, ¹ and it has been recommended that every patient with a COVID-19-related intensive care unit (ICU) stay be followed by an otolaryngologist following discharge. ² While commonly reported disorders range from true vocal fold edema and paresis to posterior glottic or subglottic stenosis, ¹ emerging reports have described tracheoesophageal fistula (TEF) in the most severe cases.^2,3 Nonmalignant TEFs have long represented a surgical challenge, with recurrences common and the management of associated tracheomalacia or stenosis often requiring additional management. ⁴ We present a case of delayed diagnosis of a COVID-19-related TEF successfully managed with multidisciplinary surgical management.

Case Presentation

A 63-year-old female with a past medical history of hypertension and morbid obesity was admitted to an outside hospital with COVID pneumonia and was subsequently diagnosed with a TEF during endoscopic replacement of a dislodged gastrostomy tube. The patient was previously treated for COVID pneumonia with high-dose steroids and subsequently required endotracheal intubation. She had undergone percutaneous tracheostomy and gastrostomy tube placement following 14 days of orotracheal intubation. Her ICU course was prolonged and complicated by gastric perforation and empyema.

During esophagogastroduodenoscopy (EGD) for gastrostomy tube replacement 6 weeks following admission, a TEF was noted 16 cm from the incisors by the gastroenterology team at an outside facility. Subsequent CT imaging further demonstrated the TEF (Figure 1a) and the patient was transferred to our institution for further management. Operative endoscopic evaluation demonstrated a defect in the posterior tracheal wall, estimated to be 3 cm in size, at the level of the tracheotomy cuff (Figure 1b). Findings also included exposed posterior cricoid plate with overlying granulation tissue, and significant anterior-posterior tracheal collapse at the level of the stoma. Esophagoscopy further demonstrated the defect, as easily evidenced by the visualization of the tracheostomy tube balloon protruding through the defect into the esophagus (Figure 1c). An image captured of the shared wall dividing distal trachea and esophagus at the inferior aspect of the defect is depicted in Figure 1d.

Figure 1.

(a) CT imaging demonstrating a TEF. A nasogastric feeding tube within the esophageal lumen allows the connection to the trachea to be appreciated. (b) Intraoperative image captured during tracheoscopy with tracheostomy tube removed, showing fistula at the site of the tracheostomy tube balloon. (c) Intraoperative image captured during esophagoscopy with the cuff of the endotracheal tube visible protruding from the trachea through the defect into the esophagus. (d) Intraoperative image captured during esophagoscopy showing the shared wall between the esophagus and trachea at the inferior aspect of the TEF.

Due to significant deconditioning from a prolonged ICU course, repair was delayed 14 months from the initial diagnosis. During this time, a cuffed tracheostomy tube was positioned 1 to 2 cm above the carina with the top of the cuff placed distal to the inferior edge of the TEF for ventilation, and nutrition was maintained primarily by tube feeds through a post-pyloric small bowel feeding tube after unsuccessful gastrostomy tube replacement. Repeated rigid bronchoscopy and esophagoscopy at the time of repair demonstrated a stable defect at the party wall of the trachea and esophagus, distal to the innominate artery. An apron-type cervical incision encompassing the tracheocutaneous fistula was used. The upper aerodigestive tract was skeletonized, and the recurrent laryngeal nerve was identified bilaterally. A median sternotomy was performed by the cardiothoracic surgery team and the innominate artery and vein were skeletonized, allowing for caudal retraction and identification of the fistula. The fistula was incised, and the esophageal defect was repaired primarily using a 3-0 V-loc barbed suture (Medtronic, Minneapolis, MN, USA). A 3 cm portion of malacic trachea, distal to the existing tracheostomy stoma, was then resected. Blunt anterior tracheal dissection to the carina allowed for tension-free tracheal closure. A pectoralis muscle flap was then raised and tunneled over the clavicle. Muscle was interposed between the tracheal and esophageal repair and secured to the contralateral tracheal rings. The tracheostomy stoma was not closed, and a tracheostomy tube sized to extend distally to the anastomosis was replaced at the conclusion of the procedure.

The patient tolerated the procedure well and was discharged from the hospital to her prior skilled nursing facility at 7 days post-op. At 3 weeks post-op, flexible tracheoscopy demonstrated a well-healing tracheal anastomosis (Figure 2a) and esophagram demonstrated no contrast extravasation (Figure 2b). Diet was advanced. At 7 weeks post-op, the patient was tolerating capping trials and her tracheostomy was decannulated. At that time, she was tolerating all nutrition by mouth with the gastrostomy tube removed. Subsequent esophagoscopy at 5 months post-op by the gastroenterology team, performed while addressing a persistent gastrocuteneous fistula, demonstrated no esophageal abnormalities.

Figure 2.

(a) Image captured during tracheal endoscopy, demonstrating the well-healing tracheal anastomosis 3 weeks following repair of TEF. (b) Still captures from modified barium swallow performed 3 weeks after repair of the TEF, showing no leak of ingested contrast material in the esophagus.

Discussion

Aerodigestive injuries as a result of prolonged intubation have become increasingly prevalent in the setting of the COVID-19 pandemic, when many patients develop severe pneumonia necessitating mechanical ventilation. In 2020, experts of the Laryngotracheal Stenosis Committee of the European Laryngological Society published an opinion piece alerting practitioners to maintain a high level of suspicion for airway-related complications among those recovering from critical COVID-19 illness suggesting that “every patient with a history of COVID-19-related ICU stay should be followed after discharge by an otolaryngologist or other airway specialist to proactively diagnose early complications at the level of the larynx and trachea.” ² In a large literature review, including 393 patients with post-acute laryngeal injuries and dysfunctions following COVID-19 infection, data suggested that the prevalence of laryngotracheal complications may reach 16% to 65%. ¹ In a more recent prospective study, follow-up endoscopic evaluation of 96 patients with a history of orotracheal intubation due to COVID-19 revealed evidence of laryngotracheal lesions in 38 (40%) patients. ⁵ The most common complication among published data is laryngotracheal stenosis (posterior glottic, subglottic, or tracheal), followed by vocal fold immobility, muscle tension dysphonia, laryngotracheal reflux, granulomas, and less commonly TEFs.^1,5,6

Nonmalignant TEFs are most commonly a result of prolonged mechanical ventilation, but the data remain inconclusive on whether they occur in greater incidence in patients with COVID-19 infection compared to patients without. ¹ However, laryngotracheal injuries such as TEF can likely be attributed to prone positioning, which increases endotracheal cuff pressure on the tracheal wall, a prothrombic state of infection leading to ischemic injury, and viral replication in the tracheal mucosa weakening the wall. ³ Concurrent use of steroids and/or nasogastric tube feeding, as required in our patient, may further increase risk.

In general, benign TEFs of any etiology are most commonly accessed for repair via cervical incision, transverse cervical collar more often than lateral cervical incision, and only rarely require an upper or median sternotomy.^4,7 Since 1991, repair via tracheal resection and anastomoses with primary esophageal closure using 2-layer technique has been adopted into favor over direct tracheal and esophageal closure without resection. ⁷ Other less favorable techniques that have been reported in the literature include esophageal reconstruction with full thickness skin graft, cervical esophagostomy, or esophageal diversion/esophagocolonoplasty.^4,6,7,8 TEFs commonly occur at the cervicothoracic junction, ⁹ and in some cases a sternotomy may be necessary to improve exposure of the carina.

Tracheal resection, particularly to remove stenotic or malacic trachea, has grown in preference for many reasons. Removal of the diseased trachea and opening of the airway offer complete exposure posteriorly to the esophageal defect, allowing for more safe and precise surgical debridement and two-layer closure. After esophageal closure, the trachea is anastomosed with sutures, while laryngeal release is done as necessary to avoid excessive anastomotic tension. This technique has proven to offer clinical benefit without increasing morbidity. ⁷ Typically, once the defect is excised and closed, a muscle flap is used to separate and reinforce the airway and esophagus to protect suture lines and prevent complications including stenosis or fistula recurrence. ¹⁰ The pedicled strap muscle flap is utilized most often, with the sternocleidomastoid a popular alternative.^4,8 The pectoralis major muscle flap is a reliable option for reconstruction throughout the head and neck^11–13 but its reported use is limited for COVID-19 patients and TEF.

Surgical repair is complicated in the case of COVID-19-associated TEF, as underlying deconditioning and malnutrition may necessitate staged repair.^9,10 One large case series demonstrated the difficulty in management of TEFs following COVID-19 pneumonia, with 6/14 (42.8%) patients expiring from infectious complications after endoscopic treatment, ⁹ emphasizing the importance for prompt identification and intervention. Spontaneous closure of TEF is rare but may occur, as demonstrated in a case of a COVID-19 patient who declined surgery but recovered with only supportive treatment with a jejunostomy tube and discontinuation of all oral feeding. ¹⁴ Success has also been shown in 4 cases of TEF in COVID-19 patients not stable enough for surgery, with one-time esophageal endoluminal suture, downstream tracheostomy cannula replacement, and percutaneous endoscopic gastrojejunostomy (PEG-J) placement, all performed at bedside. ¹⁵ Due to critical illness, other patients were managed endoscopically^9,16 or expired prior to a planned repair. ¹⁷ However, open surgical correction remains the preferred method of management in patients who are willing and able to tolerate the procedure. Specific to the repair of TEF as a complication of COVID-19, several cases describe the cervical incision approach to access and perform tracheal resection and anastomosis, primary esophageal closure, and a sternocleidomastoid muscle flap to support suture lines.^16,18 One case describes a left lateral cervicotomy with primary esophageal and tracheal repair, and sternocleidomastoid muscle flap. ¹⁹

In our patient, the distal location and large diameter of the defect necessitated a unique repair strategy. This case provides additional evidence of the efficacy of sternotomy and tracheal resection with anastomosis and esophageal closure for management of complicated TEFs. If complicated by a distal location in the esophagus or a large diameter, the pectoralis major myofascial flap is useful to consider in the repair for improved structural support and reduced risk of complications such as recurrence or airway stenosis. This multidisciplinary surgical strategy proved successful in our patient who continues to recover without complication and should be considered for future patients suffering this uncommon complication of COVID-19.

Our literature review also raises awareness for the high prevalence of all aerodigestive injuries secondary to mechanical ventilation in COVID-19. In such patients requiring prolonged intubation, it is important to avoid excessive prone positioning, monitor endotracheal cuff pressures, and use steroids sparingly. Especially in patients also requiring a nasogastric tube, it is important to maintain suspicion and observe for early signs of development of a TEF as this complication is associated with high morbidity and mortality.

Conclusion

We describe the successful surgical management of TEF secondary to COVID pneumonia. Our approach of concurrent tracheal resection, esophageal reconstruction, and pectoralis muscle onlay proved successful and is likely to be an option for similar lesions which may become more common as post-COVID-19 aerodigestive injuries become commonplace.