Role of Percutaneous Trans-Tracheal Endoscopic Approach in Managing Thoracic Tracheal Pathologies: A Step Back for Better Outcomes

Abstract

Thoracic tracheal tumors are relatively rare in routine clinical practice. Though surgery is the preferred management choice, acute presentation is challenging for the surgeon and the anesthetist. In this study, we discuss the role of the percutaneous trans-tracheal endoscopic approach (PTTEA) in the modern era of minimally invasive surgery. Its indications, advantages, complications, and anesthetic options are enumerated. A retrospective review was performed of patients with thoracic tracheal lesions treated in our department between 2015 and 2022. A total of 16 patients underwent PTTEA in an emergency after a failed fiberoptic or transoral approach and were included in the review. Twelve patients were ventilated by intermittent apnea, and four had a Cardiopulmonary bypass. Amongst the 16 patients, nine had benign, and seven had malignant pathologies. All the benign lesions were excised completely, and patients were de-cannulated, except for one case with disseminated respiratory papillomatosis. Malignant lesions underwent debulking of the lesions for acute respiratory distress, followed by initiation of adjuvant therapy. PTTEA is an effective alternative to traditional fiberoptic or rigid bronchoscopy techniques for selected patients for whom minimally invasive techniques fail. The advantages of the approach include better surgical access, hemostasis, and airway control, resulting in reduced operative time and minimal complications.

Key Points

• The acute presentation of thoracic tracheal lesions with severe respiratory distress is challenging for the surgeon and the anesthetist.

• Thoracic tracheal pathologies are usually amendable to fiberoptic or rigid bronchoscopic approaches for excision or tissue diagnosis.

• In regions of India where beetle nut chewing and oral tobacco addiction are common, trismus is encountered frequently due to oral submucosal fibrosis, which may affect any approaches through the oral cavity.

• PTTEA for thoracic tracheal pathologies is an effective alternative when traditional minimally invasive surgical techniques fail.

Introduction

Isolated tracheal tumors are rare and account for 2.6/1,000,000 newly diagnosed cancers [1]. Most of the primary lesions are malignant. Squamous cell carcinoma is the most common malignant pathology encountered, followed by adenoid cystic carcinoma [2]. Usually, patients present with nonspecific pulmonary symptoms like wheezing or cough, which delay the diagnosis and lead to an acute presentation like stridor or hemoptysis [3]. Such acute presentation in an emergency mandates rapid intervention to secure the airway. Surgery is usually the definitive management option with the addition of adjuvant therapies based on histopathology. In cervical tracheal malignancies, transcervical tracheal resection and anastomosis help to restore the airway [4]. Thoracic tracheal pathologies are usually amendable to fiberoptic or rigid bronchoscopic approaches for excision or tissue diagnosis. However, in certain situations where the fiberoptic or transoral approach fails or in a set-up lacking access to instrumentation, the percutaneous trans-tracheal endoscopic approach (PTTEA) can be lifesaving. As per our literature review, this is the largest reported series of PTTEA.

Methodology

A retrospective review was performed for all the thoracic tracheal lesions treated in our department from 2015 to 2022. The ethical board of the institute reviewed the study. A total of 16 patients who underwent PTTEA were included in the review (Table 1). All the patients were referred by our Pulmonary medicine colleagues and underwent a trial of fiberoptic-guided management. Only patients with failed fiberoptic or trans-oral approaches were taken up for PTTEA. Two patients had pre-existing tracheal access due to an indwelling Montgomery tube, with the obstruction being distal to the lower limb of the tube. Radiological images of the few patients are given in Fig. 1a and d.

Table 1.

Shows details of the patients, indications for percutaneous transtracheal endoscopic approach, the preferred mode of anesthesia, complications, and outcomes

No	Age/sex	Diagnosis	Indications	Treatment	Anesthesia	Remarks/Outcomes
1	16/M	Rosai Dorfman disease	Recurrent lesion after 3 months of fiberoptic debulking of the lesion and being treated as Rhinoscleroma, failed treatment with antibiotics from elsewhere. Our histopathology confirmed Rosai Dorfman disease. Grade 3 respiratory distress(6), morbid obesity, and intermittent bouts of hemoptysis impairing vision.	Complete excision + patient responded to steroids and methotrexate treatment for 12 months.	Cardiopulmonary bypass, which was converted to Intermittent apnea (due to Intraoperative North-South syndrome)	Decannulated after 1 year, following a negative PET scan. On disease-free follow-up for 3years.
2	28/M	Rhinosporidiosis	Grade 3 respiratory distress(6), with significant hemoptysis. Fiberoptic procedure was abandoned due to bleeding and worsening of distress.	Complete excision with cauterization of base	Intermittent apnea	Decannulated On disease-free follow-up for 18 months.
3	36/M	Rhinosporidiosis	Grade 2 respiratory distress(6). Fiberoptic procedure was abandoned due to uncontrolled bleeding.	Complete excision with cauterization of base	Intermittent apnea	Decannulated On disease-free follow-up for 3 years
4	33/M	Adenoid cystic carcinoma	Severe airway obstructive symptoms (Grade 3)(6) with grade 3 trismus (7) (Oral tobacco chewing).	Debulking + adjuvant chemotherapy + radiotherapy	Cardiopulmonary bypass	Intraoperative air embolism with ischemic stroke. On palliative care after completion of radiation therapy, with stable disease and tracheostomy in situ for 1 year
5	49/F	Adenoid cystic carcinoma	Atelectasis of one lung and emphysema of the other lung, with grade 3 distress(6); grade 3 trismus(7) due to tobacco chewing. Previous fiberoptic guided biopsy was twice negative.	Debulking + adjuvant chemotherapy + radiotherapy	Cardiopulmonary bypass	Stable disease with tracheostomy in situ for 21 months
6	55/F	Adenoid cystic carcinoma	Delayed presentation with respiratory distress grade 4(6) due to COVID lockdown. Urgent airway clearance required due to sudden deterioration during fiberoptic procedure.	Debulking + adjuvant chemotherapy + radiotherapy	Intermittent apnea	Decannulated. disease-free follow-up for 1 year.
7	57/M	Adenoid cystic carcinoma	Grade 3 trismus(7) with worsening respiratory distress, Grade 3(6). Patient was unable to cooperate for fiberoptic procedure.	Debulking + adjuvant chemotherapy + radiotherapy	Cardiopulmonary bypass	On palliative treatment
8	63/M	Squamous cell carcinoma	Acute bleeding post-Fiberoptic guided biopsy with worsening distress.	Debulking + adjuvant chemotherapy + radiotherapy	Intermittent apnea	On palliative treatment
9	63/M	Squamous cell carcinoma	Delayed presentation with grade 4 respiratory distress(6) and severe hemoptysis due to COVID lockdown. The bleeding prevented minimally invasive technique.	Debulking	Intermittent apnea	Died on the second postoperative day due to cardiac arrest.
10	46/M	Squamous cell carcinoma	Grade 3 trismus(7) and respiratory distress(6). Fiberoptic guided biopsy was negative with progressive disease.	Debulking + adjuvant chemotherapy + radiotherapy	Intermittent apnea	Stable disease on palliative care with tracheostomy in situ
11	40/F	Papilloma	Grade 2 trismus(7) with grade 3 respiratory distress(6). Fiberoptic procedure was abandoned due to worsening distress and bleeding.	Complete excision	Intermittent apnea	Decannulated On follow-up for 5 years
12	14/F	Montgomery Tube induced Tracheal stenosis	Pre-existing tracheal stoma. Presented with progressive respiratory distress.	Release of stenosis (Coblation) + Dilatation + Mitomycin-C application	Intermittent apnea	Decannulated On follow-up for 6 months
13	16/F	Montgomery Tube induced Tracheal stenosis	Pre-existing tracheal stoma. Presented with progressive respiratory distress	Release of stenosis + balloon dilatation + Mitomycin-C application	Intermittent apnea	Decannulated On follow-up for 7 years
14	36/ F	Tracheal Rhinoscleroma	Morbid obesity with worsening grade 3 distress(6). Prior 2 biopsies were not confirmatory.	Complete excision with 8 months antibiotic treatment.	Intermittent apnea	Decannulated. On disease free follow-up for 6 years.
15	35/M	Reparative granuloma	Grade 2 distress(6) and grade 2 trismus(7). Fiberoptic procedure was abandoned as the mass was firm to hard in consistency and could not be excised.	Complete excision	Intermittent apnea	Decannulated On follow-up for 8 months.
16	43/F	Recurrent Respiratory papillomatosis	Fiberoptic procedure was abandoned due to diffuse disease and uncontrolled bleeding with worsening distress (grade 3)	Multiple staged debridement	Intermittent apnea	On follow-up with tracheostomy tube in situ.

Fig. 1.

a Shows Computed Tomography (CT) of the thorax axial section with irregular soft tissue density lesion with near total obstruction of thoracic trachea. b shows Chest X-ray with soft tissue opacity in the thoracic trachea. c & d show CT with the 3-D reconstruction(1c) of the thorax with soft tissue density lesion in the thoracic trachea with an extraluminal invasion of the tracheal wall

Ventilation

All our patients were ventilated through intermittent apnea or a peripheral cardiopulmonary bypass/extracorporeal membrane oxygenation (ECMO). Under local anesthesia, venous and arterial cannulation was done and connected to the ECMO machine. Once the perfusionist confirmed the position and efficiency, the patient was administered intravenous anesthetic agents. A tracheostomy was done after infusing intravenous anesthesia. Intermittent apnea is a good option in case of non-availability of ECMO facilities or if the patient cannot undergo the cannula placement due to severe acute respiratory distress.

In patients with intermittent apnea technique, tracheostomy was first done under local anesthesia.

Jet ventilation was avoided in our series due to the risk of technical failures, the chance of distal spread of the tumor, hemodynamic instability, pneumothorax, and cervical emphysema [5].

Technique

Local infiltration was given after positioning the patient with minimal extension, or if the patient cannot tolerate the supine position, a 45-degree head upright position. A 4–5 cm cervical horizontal incision was made approximately one to two-finger breadth above the suprasternal notch corresponding to the lower level of the cervical trachea (Position sometimes varied due to the low anatomical position of cricoid cartilage). Dissection was done, and the trachea was delineated. The Isthmus of the thyroid gland was divided if required. Soft tissues and strap muscles were retracted laterally, creating a stoma. Stay sutures were used to maintain the tracheostomy stoma in position. The primary surgeon was positioned at the head end of the patient, and the assistant was on the left side. A rigid bronchoscope was not used to avoid injury to the tracheal wall, and a zero-degree nasal endoscope/telescope was utilized to visualize the lesions (Fig. 2a-d). The procedure was performed under direct vision with a three-handed technique for instrumentation and suctioning. This allowed for better visualization and a wider range of instrumentation.

Fig. 2.

Endoscopic images of different tracheal pathologies. a-Rosai Dorfman disease, b-Adenoid cystic carcinoma of the trachea, c-Rhinosporiodosis of the trachea, d-Squamous cell carcinoma of the trachea

In patients with short neck, the rigid endoscope was inserted at an angulation through the right side of the patient to avoid overlapping with the jaw. The fiberoptic was not used as the rigid endoscope provided better vision and allowed the surgeon to use both hands for surgery due to the shorter working distance.

The lesion was debulked with emphasis on hemostasis using a laryngeal wand of coblation or CO2 laser fiber or diathermy cautery. If a CO2 laser was used, FiO2 was maintained at less than 30%. Patients not undergoing ECMO were breathing spontaneously and maintained deep sedation during the initial part of the procedure until a small-sized endotracheal tube was passed distal to the obstruction after debulking to secure the airway. Total intravenous anesthesia was administered after that. A small-sized cuffed endotracheal tube (Size 5/6) was negotiated beyond the debulked lesion; if the bronchus was also involved, the tube was directed to the bronchus with a relatively patent lumen. Endoscopic debulking/ complete excision of the lesion was done based on the type of pathology encountered. Peripheral cardiopulmonary bypass was terminated after surgery. A tracheostomy tube was placed after surgery in all patients. All the patients had tracheostomy tube placed after the procedure and were ventilated overnight. The patients were administered intravenous Dexamethasone 8 mg thrice daily to avoid airway edema. The following day, a fiberoptic evaluation was performed through the tracheostomy tube to rule out any bleeding or blockage, and then the patient was weaned off the ventilator.

Patients who underwent complete excision of the benign pathology were decannulated uneventfully with a minimum follow-up of six months, except in one case of disseminated papillomatosis. Patients with malignant lesions were maintained on tracheostomy and started on adjunctive therapy. Decannulation was delayed in malignant etiologies until the completion of adjuvant treatment.

A pictorial representation of our operation room set-up of PTTEA for thoracic tracheal lesions is given in Fig. 3.

Fig. 3.

Shows a pictorial representation of the operation room set-up

Results

In our study, we reviewed 16 patients, 9(56%) males and 7(44%) females. The age range was 14–63 years. The pathologies encountered were 7(44%) malignant and 9(56%) benign; two were thoracic tracheal stenosis due to foreign body reaction/mucosal trauma in the lower end of the Montgomery tube in previously stented cases of cervical tracheal stenosis. All patients presented with acute airway obstructive symptoms to the emergency department, and one patient had distress and significant hemoptysis post fiberoptic guided biopsy.

In recurrent respiratory papillomatosis (RRP), staged debridement was performed to remove the disease. All patients with benign disease are under close follow-up with no clinical or radiologic recurrence. Eight patients have been decannulated successfully. Patients with malignant etiologies underwent debulking to relieve the acute airway obstruction and then initiated adjuvant treatment based on histopathology and disease extent. One patient had air embolism during cardiopulmonary bypass, which caused ischemic stroke, and is currently in palliative treatment. One patient died on the second postoperative day due to probable secondary bleeding and cardiac arrest. Details of all patients are tabulated in Table 1.

Discussion

Primary tracheal tumors are rare, and thoracic tracheal involvement is less common than cervical tracheal involvement. Most tracheal tumors are malignant. Since they initially present with nonspecific pulmonary complaints, many patients were treated for Asthma/COPD/Bronchitis/Tuberculosis and referred to tertiary care centers in a late stage with acute airway obstruction. In our retrospective analysis, some patients, especially those from the rural sector, presented late, and some showed late due to COVID-19 restrictions. Those who presented early with stable airways underwent transoral rigid bronchoscopy/ fiberoptic bronchoscopy/ trans-thoracic approach as per the institute protocol.

In regions of India where betel nut chewing and oral tobacco addiction are common, trismus due to OSMF (Oral submucosal fibrosis) is encountered frequently. In our series, all patients had respiratory distress (grade 2–4) [6] due to airway obstruction; one patient had hemoptysis with airway obstruction due to bleeding from the tumor. Six patients had grade 2/3 trismus. Difficult airway due to obesity, short neck, etc., were seen in three patients. In four patients, the fiberoptic procedure was abandoned due to failure in view of uncontrolled bleeding/worsening of respiratory distress. Two patients already had a tracheostomy tube in situ with respiratory distress due to distal segment stenosis. In these situations, instrumentation becomes challenging and, in some patients, even impossible. Lesions like Rhinosporidiosis are very vascular and challenging to control with minimally invasive techniques. Patients with high respiratory distress could not cooperate during fiberoptic bronchoscopy. Some patients deteriorated during the procedure, warranting urgent airway clearance and securing ventilation as a lifesaving measure.

The advantages of PTTEA include easy accessibility, good control of bleeding (Diathermy and better suctioning), and better airway control with decreased risk of aspiration. The better visualization and instrumentation with the three-hand technique is a significant added advantage. In a previously tracheostomized patient, as in our study, PTTEA will be the best option considering the above benefits. The ready availability of instruments in an existing ENT set-up decreases the response time treatment cost and allows secondary care centers to provide this lifesaving care early.

The anesthetic management in lower tracheal airway lesions is challenging. ECMO is considered a safe and secure ventilation option. There are two types of extracorporeal support – Veno-venous (VV) and Veno-arterial (VA). In VV-ECMO, the deoxygenated blood is taken from the SVC and returned to the heart as oxygenated blood through IVC. In contrast, in VA-ECMO, the deoxygenated blood is taken from the vein and returned through the artery to the heart. Under local anesthesia, venous and arterial cannulation is done and connected to the ECMO machine. The patient is administered intravenous anesthetic agents once the perfusionist confirms the position and efficiency of ECMO. Intermittent apnea is a good option in case of non-availability of ECMO facilities or if the patient cannot undergo the cannula placement due to severe worsening acute respiratory distress. Different modes of ventilation could be intermittent apnea, rigid bronchoscope, jet ventilation, or Peripheral cardiopulmonary bypass. Our study used intermittent ventilation in 12 patients and peripheral cardiopulmonary bypass in 4 patients. Unfortunately, in one patient with Veno-venous (femoro-femoral) ECMO, we encountered Harlequin syndrome, a north-south syndrome [7], where we converted it into an intermittent apnea method. This happens in patients with good left ventricular function. In this patient, when anesthesia was induced with propofol and fentanyl, a watershed zone was created, with the upper body getting deoxygenated blood from the left ventricle and the lower body oxygenated from the extracorporeal circuit, which was complicated more by morbid obesity, causing decreased functional residual lung capacity.

We successfully used Veno-Arterial ECMO(Axillo-femoral) in the other three patients. In whom ECMO couldn’t be arranged, as the distress was very acute or the set-up was unavailable, we proceeded with intermittent ventilation and apnea technique.

A literature search on the management of thoracic tracheal lesions by PTTEA published in English literature till 1st December 2022 was performed using keywords tracheal tumors, tracheal obstruction, thoracic tracheal lesions, endoscopic trans tracheal approach, rigid bronchoscopy, stridor, ECMO in Medline.

The initial study was published by Sharma et al. [8] in 2018 on a patient with tracheal glomus under endotracheal intubation with no complications. It was followed by a few more case reports by S. Reddy et al. [9] and K. Aggarwal et al. [10] on inflammatory myofibroblastic tumors. A couple of case series with three patients each by Khaund G. et al. [11] and Sathe et al. [12] could also be found, with one intraoperative death reported in the later study. In comparison, our study included 16 patients, which is relatively higher than the previous studies with some unreported pathologies, ventilatory procedures, and complications.

A study by S. Misra et al. [7] reported the excision of thoracic tracheal lesions using peripheral cardiopulmonary bypass. According to our search, no other study in literature used the same. Cardiopulmonary bypass is very effective as the airway can be isolated without requiring an endotracheal tube for ventilation. Thus, adequate space and time are available for the surgery. As there is no sharing of the airway, the instrumentation becomes easy, and the chance of airway fire with a laser is completely negated.

Though complications like aspiration pneumonia and tracheal wall injuries are reported in the literature, we haven’t encountered any. However, we had complications like intra-operative Harlequin syndrome and cerebrovascular accident due to air embolism. Unfortunately, one patient died due to cardiac arrest on the second postoperative day.

The limitation of the study is that it has a very small sample size; thus, no universal applicability can be speculated based on the findings. Limitations of the technique are that a tracheostomy is required, which is associated with morbidity to the patient.

Conclusion

PTTEA for thoracic tracheal pathologies is an effective alternative when traditional minimally invasive surgical techniques fail. It has the advantage of better accessibility, visualization, hemostasis, and airway control with decreased risk of aspiration. The 3-hand technique helps in better instrumentation and tumor handling, with early restoration of the airway. Vascular pathologies like Rhinosporidiosis, high grade of respiratory distress with a history of hemoptysis, and high grade of trismus are common instances where minimally invasive techniques can fail, warranting PTTEA as a lifesaving measure. Peripheral cardio-pulmonary bypass, if available, is the preferred anesthetic approach.

Declarations

Ethical Approval

Ethical clearance obtained from the Institutional Ethics Committee, AIIMS Bhubaneswar.

Financial Disclosure

The study was not funded by any organization or Institution.

Conflict of Interest

There are no conflicts of interest among the authors.