A 21-year-old woman, receiving the insertion of one covered Y-shaped SEMS for the treatment of complex PTTS 2 years ago, was admitted to our department with a complaint of dyspnea and frequent choking with water for 2 weeks. The dyspnea was noticed when walking on flat ground or climbing one flight of stairs. She also had sputum, but no fever, night sweats, or weight loss. She had a history of pulmonary TB in 2004, which was cured following the four-drug regimen (2HRZE/10HRE) treatment. The previous bronchoscope had established the diagnosis of complex PTTS with varying degrees of malacia in multiple locations (middle and lower trachea; right and left bronchus), associated with severe obstructive ventilatory impairment. She had repeated respiratory symptoms even after the numerous airway intervention, such as electrocautery, balloon dilatation, and cryotherapy.
She had a temperature of 36.3 C, a heart rate of 112 beats per minute, a breath of 21 times per minute, and a blood pressure of 95/76 mmHg. The physical examination revealed stridor on the anterior neck and rales in the bilateral lower lungs on admission; the rest were insignificant. Laboratory tests showed that leukocyte count was 11.29 x 109/L and neutrophil percentage 81.4%, and the others were unremarkable. The chest computed tomography (CT) demonstrated severe granulation tissue hyperplasia on the upper edge of the covered SEMS, inducing the shearing force against the anterior esophageal wall (Figure 1A), and a part of the stent penetrated the esophagus (Figure 1B). The bronchoscopy confirmed severe granulation tissue hyperplasia on the upper edge of the covered SEMS, causing 90% airway obstruction (Figure 1C). We made radical incisions toward the granulation tissue with electrocautery, followed by mechanical debulking with the forceps and flexible cryoprobe. After eradicating the granulation tissue, the TEF was located in the 7 o'clock position (Figure 1D).
The patient was diagnosed with stent-related TEF and complex PTTS. She underwent percutaneous endoscopic gastrostomy for enteric feeding and artificial nutrition. She was administered anti-infective therapy with cefoperazone sulbactam and acid-suppressive treatment with ranitidine. A multidisciplinary discussion was conducted among pulmonologists, gastroenterologists, cardiothoracic surgeons, and anesthesiologists. The patient consented to remove the covered Y-shape SEMS via bronchoscopic intervention because the surgical repair was impossible and deemed high risk. Since the stent was partially embedded in the esophagus (Figure 2A), we vaporized this embedded part with the neodymium-doped yttrium aluminum garnet laser (Figure 2B). We then gently sledded the slope of the rigid scope between the stent and the airway to avoid tearing the TEF during the stent removal. The covered Y-shape SEMS was removed via rigid bronchoscopy in a piecemeal fashion (Figure 2C) under general anesthesia, revealing the TEF and complex PTTS in the trachea (Figure 2D). The whole procedure was completed in concert with cardiothoracic surgery backup.
The patient presented with worsening dyspnea and severe stridor after removing the covered Y-shape SEMS. She had a large TEF (Figure 3A), associated with complex PTTS, with the narrowest part being 5.7 mm in diameter (Figure 3B), unsuitable for surgical repair or rigid bronchoscopy (which could increase the risk of TEF perforation). The patient consented to receive the insertion of another fully covered Y-shape SEMS (Microtech Co., Ltd., Nanjing, China) to occlude the TEF orifice and establish the patency of the tracheobronchial tree (Figure 3C) on a compassionate basis. The upper edge of the stent extended more than 10 mm beyond the range of the TEF (Figure 3D). The local administration of recombinant bovine basic fibroblast growth factor (rbFGF) (Beifuji Spray, Essex Bio-Pharmaceutical Co., Ltd.; 6,000 IU/time) was sprayed into and around the fistula through the esophagus using the bronchoscope every other week as we reported (for a total of 4 times). The TEF was closed 3 months after inserting the fully covered Y-shaped SEMS and covered by epithelium and granulation tissue (Figure 3E) in endoscopy. The gastrostomy tube was removed when the patient started with normal feeding. The patient received routine bronchoscopy and removal of granulation tissue on the edges of the SEMS every 2-weeks to maintain the stent patency and reduce the risk of stent-related TEF. She showed sustained clinical and radiographic improvement in the follow-up up to 1.5 years after the treatment.