Tracheal carina reconstruction, fistula repair and esophageal reconstruction assisted by extra-corporeal membrane oxygenation for complex huge tracheoesophageal fistula: a case report

Introduction

Esophageal-respiratory fistula (ERF) is a pathological communication between the respiratory tract and the adjacent esophagus due to various factors. The fistula can occur in any part of the trachea and bilateral bronchus below the larynx, but the most common site is between the middle of the esophagus and the main bronchus. According to the etiology, ERF can be divided into two categories: benign and malignant. Benign ERF is more common in postoperative surgery, esophageal stent implantation, tracheal intubation and other respiratory tract injuries, infectious diseases (such as tuberculosis, Crohn’s disease and other esophageal granulomatous diseases, syphilis, fungal infection, etc.), trauma, etc. (1-6). Malignant ERF is usually secondary to advanced esophageal cancer, advanced lung cancer, mediastinal malignancy, thyroid cancer, and other tumors that metastasize to respiratory tract and thoracic tumors after radiotherapy. A case of benign complex tracheoesophageal fistula is reported. We completed esophageal stent removal, tracheoesophageal fistula repair, tracheal carina reconstruction partial esophageal resection, and gastro-esophageal replacement operations assisted by extra-corporeal membrane oxygenation (ECMO). We present this case in accordance with the CARE reporting checklist (available at https://acr.amegroups.com/article/view/10.21037/acr-24-14/rc).

Case presentation

A 34-year-old female patient was diagnosed with ovarian cancer in 2017 and underwent a bilateral total ovaries and uterus resection. About 2 months after surgery, bone metastasis, lung metastasis, pelvic tumor recurrence, and multiple abdominal lymph node metastasis were found. After chemotherapy, the tumor gradually shrank and disappeared. In April 2018, the tumor cell reduction operation was performed, and the postoperative R0 was achieved. One year later, brain and lung metastases were found, so she received regular chemotherapy and radiotherapy, then the metastases focus gradually shrank and disappeared after treatment. Mediastinal lymph nodes metastasis was found again in 2021. The patient received local chest radiotherapy combined targeted therapy again. After that, the patient began to develop progressive dysphagia and eating obstruction with the diagnosis as radiation esophagitis (Figure 1). The patient underwent endoscopic balloon dilation twice within 2 months (Figures 2,3). Unfortunately, the postoperative effect was not satisfactory along with dysphagia occurred again 2 months later, then esophageal stent implantation was performed (Figure 4). After the implantation of esophageal stent, the patient’s dysphagia was effectively alleviated. However, after the operation, she developed increasingly severe cough and sputum until she was admitted to hospital due to dyspnea. She was diagnosed as tracheoesophageal fistula combining with serious pulmonary infection and atelectasis of the lower lobe of the left lung due to the compression of the stent (Figure 5).

Figure 1 Radiation esophagitis. (A,B) Gastroscopy revealed stenosis of the middle esophagus. (C) Upper gastrointestinal angiography revealed stenosis of the middle esophagus.

Figure 2 The first-time endoscopic balloon dilation. (A) Gastroscopy revealed esophageal stenosis at a distance of 27 cm from the incisor. (B) In operation. (C) Post-operation.

Figure 3 The second-time endoscopic balloon dilation. (A) Gastroscopy revealed esophageal stenosis at a distance of 27 cm from the incisor. (B) In operation. (C) Post-operation.

Figure 4 Esophageal stent implantation. (A) Chest X-ray after esophageal stent implantation. (B) Gastroscopy revealed the esophagus was smooth after esophageal stent implantation. (C) Gastroscopy revealed the upper margin of esophageal stent after implantation.

Figure 5 Tracheoesophageal fistula combining with pulmonary infection and atelectasis of the lower lobe of the left lung. (A) Preoperative computerized tomography revealed left atelectasis. (B) Preoperative computerized tomography revealed stenosis and occlusion of the left main bronchus. (C) Preoperative bronchoscopy revealed the left main bronchus was blocked by esophageal stent in the tracheal carina. (D) Preoperative bronchoscopy revealed external pressure stenosis in the middle and lower tracheal segment.

Prior to surgery, the patient was on spontaneous respiration with respiratory rate (RR): 24/min. Body temperature (BT), blood pressure (BP) and heart rate (HR) were normal. We provided nutrition by jejunal nutrition tube with nasal feeding enteral nutrition emulsion and liquid diet, and she had no jejunostomy or a gastrostomy. She was not septic. Because she was allergic to cephalosporins, so we started fosfomycin as an anti-infection treatment. Then we performed the surgery for her. Firstly, we connected Maquet ECMO (Kunming, China) and the whole set of pipes after pre-filling while connecting the air and oxygen mixing pipe with 100% oxygen concentration and 3 L/min air flow rate. The ECMO configuration was veno-venous (femoral vein-internal jugular vein). Confirmed the connection of pipes and the water tank with the temperature at 38 ℃. The ECMO flow rate was increased to 1.5 L/min, while the vascular clamp on all channels was disconnected, the flow rate was gradually increased and maintained to 2.5–4 L/min, the blood pressure was maintained between 89–96/50–60 mmHg. The surgical incisions were both the right thorax and median abdominal incision. We found the gastric vessels were brittle and adhered to the surrounding tissues due to a history of radiotherapy and chemotherapy. First, we dissected the stomach up to the cardia and down to the duodenum, then gastric tube was prepared. Next, we released esophagus along the front and back walls of the esophagus from the esophageal hiatus of the diaphragm to the esophageal stent while from the esophageal stenosis to the top of the chest, then we pulled gastric tube into the thorax, and the lower end of esophagus was severed at cardia and the upper end of the esophagus was severed at 3 cm from the top of the thorax. The whole layer of the esophagus was opened longitudinally, and the esophageal stent was successfully removed. A huge fistula in the left main bronchial membranous portion was visible with a length of about 5 cm after the stent was removed. The fistula extended from the left main bronchus to the top of the carina, and the structure of the tracheal membranous portion behind the carina completely disappeared, tracheal intubation was visible, and a large amount of pus was visible in the left main bronchus. Mechanical ventilation could not be performed during the operation, so ECMO assisted breathing was performed. After cleaning up the purulent secretions and necrotic tissue around the operation field, the upper and lower esophagus of the fistula was preserved, and the esophageal mucosa was cauterized with an electric knife to divide it into a long and a short muscle flap. The short muscle flap was used to cover along the structure of the carina with the left and right main bronchus, suture the muscle flap to reconstruct the carina and repair the leak of the left main bronchus. Then the long muscle flap was used to cover the short muscle flap and suture their edge, so that the carina reconstruction and the tracheal fistula orifice were covered by two layers of esophageal wall (Figure 6). Surgical procedures are showed in Figure 7. Finally, the stomach and esophagus were anastomosed end to end in the chest cavity. Sixteen days after the operation, the patient was discharged successfully (Figure 8). The patient could maintain a normal diet without anastomotic stenosis and recurrent tracheoesophageal fistula after surgery in the 8 months follow-up period of time and had no symptoms of pulmonary infection such as fever and cough. The nutritional status and the quality of life was significantly improved. The patient has now started follow-up chemotherapy.

Figure 6 Surgical procedures. (A,B) Esophageal stent removal. (C) The giant fistula was exposed and the esophageal wall was reconstructed for reinforcement suture after tracheal carina reconstruction. 1: right lung; 2: azygos vein stump; 3: main trachea; 4: right bronchus stump. (D) The giant fistula was exposed and the esophageal wall was reconstructed for reinforcement suture after tracheal carina reconstruction. 1: chest wall; 2: right lung; 3: esophagus; 4: right bronchus stump. (E) Both sides of the esophageal walls were used as patches to strengthen the fistula. 1: chest wall; 2: right lung; 3: esophagus; 4: right bronchus stump. (F) Both sides of the esophageal walls were used as patches to strengthen the fistula. Dashed square: carina covered with flaps of esophagus.

Figure 7 Surgical procedures. (I) Disconnect esophagus between the upper and lower edges of the tracheoesophageal fistula. (II) Divide the disconnected esophagus into a long and a short flap. (III) Use the short muscle flap to cover along the structure of the carina with the left and right main bronchus, suture the muscle flap. (IV) Use the long muscle flap to cover the short muscle flap and suture their edge. (V) End to end anastomose the upper margin of the tube stomach and the lower margin of thoracic esophageal in the chest cavity.

Figure 8 Post-operative computerized tomography scan.

All procedures performed in this study were in accordance with the ethical standards of the institutional and/or national research committee(s) and with the Helsinki Declaration (as revised in 2013). Written informed consent was obtained from the patient for the publication of this case report and accompanying images. A copy of the written consent is available for review by the editorial office of this journal.

Discussion

If the anastomosis cannot be finished directly after tracheal segmental resection, special types of tracheal resection, such as slide tracheoplasty, oblique resection and reconstruction, and autologous tissue flaps were preferred depending upon the site and size of the fistula (7). This is a case of extremely complex tracheoesophageal fistula. The patient was assessed by the anesthesiology department as having difficult airway before surgery. Due to the unclear airway damage caused by tracheal fistula, it may be difficult to maintain oxygenation through tracheal intubation during the operation, and ECMO extracorporeal circulation is required to support the whole operation and postoperative supportive treatment. Both single-stage esophageal reconstruction and esophageal diversion and then staged reconstruction are optional procedures for the patient, but she refused to get esophageal diversion. The preoperative difficulties we encountered were as follows: (I) the lower tracheal segment of the patient was narrow due to the compression of the esophageal stent, and the huge fistula was located at tracheal carina and the adjacent left main bronchus. When the tracheal carina was reconstructed after the esophageal stent was removed and the leak was repaired, the conventional tracheal intubation mechanical ventilation could not maintain the patient’s respiratory function during the operation. (II) How to repair the huge tracheal fistula? How to reconstruct the tracheal carina? (III) The esophageal stenosis of the patient is about 27 cm away from the incisor, which is relatively high in position. It is extremely difficult to remove the esophageal stent to repair the fistula, remove the narrow esophagus, and use stomach to replace the esophagus for functional reconstruction. (IV) Malignant tumors, radiotherapy and chemotherapy, and multiple operations lead to malnutrition and extreme weakness of the patient, resulting in a high risk of complications and death during and after surgery.

The surgeon was required not only to repair the fistula and treat the pulmonary infection for etiological treatment, but also to remove the esophageal stent and remove the narrow section of the esophagus so that the patient can have normally oral feeding. Simple tracheal repair could not be performed for the huge tracheal bulge fistula. After the reconstruction of the tracheal carina, we further used the method of “double-flaps of esophagus” to consolidate and repair the fistula, that is, the esophageal wall was used as the patch to consolidate and suture the fistula twice. From the postoperative review and follow-up, we indicate that our practice is beneficial to the patient.

Conclusions

The “double-flaps of esophagus” method can be used as an effective surgical treatment for patients with complex tracheoesophageal fistula, and can significantly improve the prognosis of patient, while ECMO extracorporeal circulation can be used to maintain oxygenation during the operation for airway damage caused by tracheal fistula complicated with serious lung infection.

Acknowledgments

We thank Dr. Manjun Chen, Dr. Yanan Bao, Dr.Yue Cui and Dr. Yong Zhou from the Department of Thoracic Surgery of The First Affiliated Hospital of Kunming Medical University for their sincere and professional support of our work.

Footnote

Reporting Checklist: The authors have completed the CARE reporting checklist. Available at https://acr.amegroups.com/article/view/10.21037/acr-24-14/rc

Peer Review File: Available at https://acr.amegroups.com/article/view/10.21037/acr-24-14/prf

Funding: This study was supported by Provincial Clinical Key Specialty of Yunnan (No. 300067).

Conflicts of Interest: All authors have completed the ICMJE uniform disclosure form (available at https://acr.amegroups.com/article/view/10.21037/acr-24-14/coif). The authors have no conflicts of interest to declare.

Ethical Statement: The authors are accountable for all aspects of the work in ensuring that questions related to the accuracy or integrity of any part of the work are appropriately investigated and resolved. All procedures performed in this study were in accordance with the ethical standards of the institutional and/or national research committee(s) and with the Helsinki Declaration (as revised in 2013). Written informed consent was obtained from the patient for the publication of this case report and accompanying images. A copy of the written consent is available for review by the editorial office of this journal.

Open Access Statement: This is an Open Access article distributed in accordance with the Creative Commons Attribution-NonCommercial-NoDerivs 4.0 International License (CC BY-NC-ND 4.0), which permits the non-commercial replication and distribution of the article with the strict proviso that no changes or edits are made and the original work is properly cited (including links to both the formal publication through the relevant DOI and the license). See: https://creativecommons.org/licenses/by-nc-nd/4.0/.

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