Operative and non-operative treatment of benign subglottic laryngotracheal stenosis *

European Journal of Cardio-thoracic Surgery 26 (2004) 818 822 www.elsevier.com/locate/ejcts Operative and non-operative treatment of benign subglottic laryngotracheal stenosis * Abstract Anna Maria Ciccone a, *, Tiziano De Giacomo b, Federico Venuta b, Mohsen Ibrahim a, Daniele Diso b, Giorgio Furio Coloni b, Erino A. Rendina a a University of Rome La Sapienza, Division of Thoracic Surgery, Sant Andrea Hospital, Via Grottarossa, 1035-1039, Rome 00189, Italy b University of Rome La Sapienza, Division of Thoracic Surgery, Policlinico Umberto I, Viale del Policlinico 150, Rome, Italy Received 15 October 2003; received in revised form 8 June 2004; accepted 9 June 2004; Available online 7 August 2004 Objective: Surgery is the first line of treatment for laryngotracheal stenosis; Montgomery tube or permanent tracheostomy have been so far the only alternatives. Nd-YAG laser resection and indwelling endotracheal stents have rarely been used in subglottic stenosis for anatomic and technical reasons. We have used the latter approach to optimize the timing of surgery or to achieve palliation without tracheostomy. Methods: Between 1991 and 2001 we have treated 18 patients with subglottic stenosis (10 males, 8 females; age range 14 78, mean 34). The upper margin of the stricture was 2 mm to 1 cm below the vocal cords; the stenotic segment extended from 1.5 to 5 cm. Three patients had tracheostomy done elsewhere. Four patients (Group I) had laser and stenting by a Dumon prosthesis as the only treatment; six had laser and stenting (#4) followed after 1 6 months by laryngotracheal resection (Group II); eight had surgery alone (Group III). Results: In Group I, one patient required repositioning of the stent and in two the stent was removed; two patients died of their underlying disease; at a follow-up of 2 9 years all living patients did well but required permanent aerosolized therapy and periodical bronchoscopy. In Group II, we had two wound infections due to airway colonization by staphylococcus aureus. In Group III, two patients developed anastomotic postoperative stenosis, treated by laser (#2) and stenting (#1), and one patient with previous tracheostomy had a wound infection. Overall, in the 14 surgical patients (Groups II and III) stenosis occurred in 14.2% and infection in 21.3%. After a follow up of 15 months to 12 years, all surgical patients breathe and speak well. Conclusions: Laser resection and endoluminal stenting can be a viable alternative to surgery or optimize the timing of operation in patients with subglottic stenosis. q 2004 Elsevier B.V. All rights reserved. Keywords: Benign laryngotracheal stenosis; Postintubation tracheal stenosis; Tracheal surgery 1. Introduction Benign stenosis involving the subglottic region represents a major therapeutic challenge [1 3]. Surgery is the first line of treatment for laryngotracheal stenosis and leads to high rate of success [4 8]. Although the preservation of the recurrent laryngeal nerves remains one of the most * Presented at the joint 17th Annual Meeting of the European Association for Cardio-thoracic Surgery and the 11th Annual Meeting of the European Society of Thoracic Surgeons, Vienna, Austria, October 12 15, 2003. * Corresponding author. Tel.: C39-06-80345-773; fax: C39-06-8034-5003. E-mail address: amciccone@hotmail.com (A.M. Ciccone). vexing problems, a number of reports show good results with respect to technical problems [3 6,9 11]. Montgomery tube or permanent tracheostomy have been so far the only alternatives to surgery. In recent years, the interest for endoscopic treatment modalities, especially laser, has increased [12 14] in tracheal surgery; laser-assisted endoscopy (with or without stenting) has however rarely been used in subglottic stenosis for anatomical and technical reasons [15]. We have employed the latter treatment modality in severely compromised patients or to allow stabilization of the stenosis. Patients were either converted to surgery when the stenosis was stabilized, or offered an acceptable palliation of symptoms if surgery was not feasible for their compromised general health. 1010-7940/$ - see front matter q 2004 Elsevier B.V. All rights reserved. doi:10.1016/j.ejcts.2004.06.020

A.M. Ciccone et al. / European Journal of Cardio-thoracic Surgery 26 (2004) 818 822 819 2. Methods Between 1991 and 2001, we treated 18 patients with benign subglottic stenosis. Ten were males and 8 females with an age range of 14 78 years (median 34 years). The cause of airway stenosis was iatrogenic in 16 patients who had undergone intubation for cardiorespiratory resuscitation. The main causes of intubation are reported in Table 1. Two stenoses were idiopathic. Preoperative assessment included accurate laryngotracheal endoscopic examination to assess the grade of inflammation and oedema of the trachea, especially at the margins of the stenosis, the presence of tracheomalacia, the integrity of the vocal cords, and the extention of tracheal involvement. Neck and chest CT scan and, in the recent years, spiral CT scan were also performed to determine the extention of stenosis; radiologic imaging also allowed to obtain more information on the extraluminal region. The upper margin of the stricture was 2 mm to 1 cm. below the vocal cords; the stenotic segment extended from 1.5 to 5 cm. Three patients had tracheostomy done elsewhere. We subdivided all patients in three groups. In Group I, 4 patients were managed by laser and stenting by a silicon prosthesis as the only treatment. These were patients with contraindications to open surgery because of poor general status or pulmonary or cardiac disease. In Group II, 6 patients underwent laryngotracheal resection after laser treatment (all 6 patients) and positioning of a silicon stent (4 patients). This strategy was followed to allow the stenosis to stabilize without tracheostomy or to improve compromised general status. Surgery was performed after 1 6 months, when the stenosis appared stabilized after endoscopic reevaluation. In Group III, 8 patients with stabilized stenosis and good general health underwent surgery as the only treatment. Three of these patients had tracheostomy done elsewhere. All the endoscopic procedures were performed with the rigid bronchoscope (Efer-Dumon rigid bronchoscope; Efer Medical, La Ciotat, Cedex, France) under deep sedation with short-acting narcotics and local anesthesia (2% xylocaine). The operative approach was through a collar incision and the laryngotracheal resection was performed on the base of the technique described by Pearson in 1975 [10]. The line of resection began at the inferior border of the thyroid cartilage anteriorly and passed below the cricothyroid joint behind, removing the anterior and lateral aspects of the cricoid arch and leaving intact the posterior cricoid plate. Table 1 Main causes of intubation for cardiorespiratory resuscitation Cause No. of patients Politrauma 10 Respiratory failure following surgery 2 Acute cardiac failure 2 Neurological problems 1 Acute respiratory failure in COPD 1 This manoeuvre allowed preservation of the recurrent laryngeal nerves. The amount of trachea resected ranged from 1.5 to 5 cm. No laryngeal release was performed, as the technique of mobilization of the trachea was sufficient to expose the stenotic lesion and to perform tracheal resection and anastomosis. After resection, the distal airway was intubated with an armoured endotracheal tube. The anastomoses were performed with interrupted sutures (3-0 absorbable monofilament material) tied on the outside. With the neck hyper extended to improve the exposure, all the sutures were placed starting from the back and were left untied. The neck was then flexed, the nasotracheal ventilation tube was readvanced distal to the anastomosis and the two ends of the airway were allowed to reach applying gentle traction simultaneously on all sutures. Sutures were then tied starting from the front. The nasotracheal tube was left in place in the awakened patient for 24 h and then withdrawn after bronchoscopic check of the anastomosis. 3. Results There was neither intraoperative nor perioperative mortality. None of the patients was lost to follow-up. No patient required postoperative tracheostomy. In Group I (4 patients), no complications were observed during and after endoscopic Nd:Yag laser treatment. The only complication related to stent placement was dislocation in one patient who required repositioning of the stent. In one patient the stent was removed after 2 years. Bronchoscopy demonstrated the absence of malacia and an airway essentially normal in diameter. Two patients died of their cardiorespiratory disease after 3.5 and 6 years with no signs Fig. 1. Sixty-nine year old lady with diabetes and chronic heart failure developing subglottic stenosis following intubation for myocardial ischemia. The stent was left in place for 3.5 years. The cricoid and the anterior commisure of the vocal cords can be seen.

820 A.M. Ciccone et al. / European Journal of Cardio-thoracic Surgery 26 (2004) 818 822 of airway stenosis. During a follow up ranging between 2 and 9 years, patients did well but required permanent aerosolized and/or steroid therapy to avoid plugging with secretions and airway oedema. These patients also require periodical bronchoscopy to assess vocal cord integrity and function, the correct position and the patency of the stent, and the status of the mucosa of the remaining airway. All of these patients showed normal voice and adequate breathing for normal activities. In Group II, 6 patients were treated before surgery with the laser and in four cases we placed an endoluminal stent (Fig. 1). The goal of avoiding tracheostomy was achieved in all patients. We did not report any complication related to laser therapy and stent positioning. Surgery was performed 1 to 6 months after laser treatment when the stenosis appeared stabilized and the stent could be removed. No intraoperative and perioperative mortality occurred. Perioperative minor complications occurred in two patients (33%) who had superficial wound infection due to airway colonization by staphylococcus, and managed with drainage, antibiotics and conservative therapy. No major complications occurred and all patients breath and speak well after 2.5 to 10 years. In Group III (Fig. 2) we had no intraoperative and perioperative mortality. Complications occurred in two patients who developed postoperative stenosis (granuloma) at the anastomotic level. Both stenoses were treated by laser vaporization and in one patient we positioned a stent. The stent was removed after 1 year and the calibre of the anastomosis was slightly inferior than the remaining portion of the trachea, but yet above 90% of normal and without functional impairment. One patient with preoperative tracheostomy had a wound infection treated with drainage and conservative therapy. Overall in the 14 surgically treated patients we observed 2 (14.2%) major complications (stenosis) and 3 (21.3%) Fig. 2. Seventy-two-year old gentleman with emphysema and chronic respiratory insufficiency. (A) Before surgery a fibrous stenosis associated with a granuloma is visible below the vocal cords at the cricoid level. (B) 1 month after surgery. (C) 6 months after surgery.

A.M. Ciccone et al. / European Journal of Cardio-thoracic Surgery 26 (2004) 818 822 821 minor complications (wound infection). After a follow-up ranging from 15 months to 12 years all surgical patients present voice and respiration completely normal. 4. Discussion Surgical management of benign tracheal and laryngotracheal stenosis is the treatment of choice and may be done with high success rate [16,17]. However, primary surgery is not always feasible. Contraindications to open surgery can be either general or local. High-risk patients with severe associated systemic or cardiac diseases, or a stenosis not yet stabilized with severe inflammatory signs of the tracheal mucosa can contraindicate primary surgery. Temporary Montgomery T-tube or permanent tracheostomy have been so far the only alternatives. However, this solution in patients who require tracheostomy for surgical contraindications has two disadvantages: the possible increase of the length of stenosis and the development of airway bacterial colonization. In order to avoid tracheostomy which would complicate surgical repair, we have treated high risk patients and patients with benign subglottic stenosis not yet stabilized, with Nd:YAG laser resection with or without indwelling endotracheal stents. This necessarily heterogeneous group of patients shows high variability in the outcomes. Some patients, whose compromised general status did not improve and were thus not suitable for surgery, had good results and quality of life after non-operative treatment, with a good voice and adequate breathing for normal activities. Other patients, endoscopically treated, required multiple laser treatments and frequent bronchoscopy, which impaired their quality of life. While this less aggressive, low risk approach is more acceptable for elderly patients, definitive surgical treatment is more appropriate for younger patients who are less likely to tolerate prolonged limitations in their lifestyle. However, patients with severe inflammatory changes must be excluded from surgery and treated only when the stenosis appears stabilized after a suitable period of observation. In fact, restoration of a healthy mucosa is mandatory to obtain good results. In this setting the endoscopic management may be particularly valuable to provide time to reduce the damage due to inflammation and oedema without tracheostomy, while awaiting definitive surgical treatment. Furthermore, in our experience the use of laser with or without stent positioning did not increase the morbidity rate and it was used as a therapeutic option without increasing surgical complications and postoperative mortality. On the other hand, patients who had wound infection after surgery were as one would predict those who received preoperative tracheostomy, thus suggesting that tracheostomy plays a role in postoperative complications. We also noticed that the fear of dislocation of a subglottic, indwelling prosthesis is overestimated; in fact, in our series, this occurred only in one patient. This was likeky due to the characteristics of the fibrous stricture, which is usually stiff and rigid and reduces tracheal calibre to that of the cricoid, thus providing superior stability for the stent. This is demonstrated also by the fact that none of the patients had vocal cord dysfunction due to prosthesis dislocation. Although open surgery is the treatment of choice for benign subglottic laryngotracheal stenosis, the operation can not be performed primarily in all patients. If local active inflammation or compromised general health contraindicate surgery, laser assisted endoscopy with stenting can represent a viable alternative. Also, the potentiality of the combined approach should be considered to achieve good palliative and definitive results without tracheostomy. References [1] Grillo HC, Mark EJ, Mathisen DJ, Wain JC. Idiopathic laryngotracheal stenosis and its management. Ann Thorac Surg 1993;56: 80 7. [2] Couraud L, Martigne C, Houdelette P, Dumas PJ, Morales F. Intérèt de la resection cricoidienne dans le traitment des sténoses cricotrachéales après intubation. Ann Chir Thorac Cardiovasc 1979;33: 242 6. [3] Grillo HC. Primary reconstruction of airway after resection of subglottic laryngeal and upper tracheal stenosis. Ann Thorac Surg 1982;33:3 18. [4] Couraud L, Jougon J, Velly JF. Surgical treatment of nontumoral stenosis of the upper airway. Ann Thorac Surg 1995;60:250 60. [5] Grillo HC, Mathisen DJ, Wain JC. Laryngotracheal resection and reconstruction for subglottic stenosis. Ann Thorac Surg 1993;53: 54 63. [6] Pearson FG, Brito-Filomeno L, Cooper JD. Experience with partial cricoid resection and thyrotracheal anastomosis. Ann Otol Rhinol Laryngol 1986;95:582 5. [7] Rea F, Callegaro D, Loy M, Zuin A, Narne S, Gobbi T, Grapeggia M, Sartori F. Benign tracheal and laryngotracheal stenosis: surgical treatment and results. Eur J Cardiothorac Surg 2002;22:352 6. [8] Couraud L, Jougon J, Velly JF, Klein C. Sténoses iatrogénes de la voie respiratoire. Evolution des indication thérapeutiques. A partir de 217 cas chirurgicaux. Ann Chir Thorac Cardiovasc 1994;12: 359 74. [9] Gerwat J, Bryce DP. The management of subglottic stenosis by resection and direct anastomosis. Laryngoscope 1974;84:940 7. [10] Pearson FG, Cooper JD, Nelems JM, Van Nostrand AWP. Primary tracheal anastomosis after resection of the cricoid cartilage with preservation of recurrent laryngeal nerves. J Thorac Cardiovasc Surg 1975;70:806 16. [11] Maddaus MA, Toth JLR, Gullane PJ, Pearson FC. Subglottic tracheal resection and synchronous laryngeal reconstruction. J Thorac Cardiovasc Surg 1992;104:1443 50. [12] Sharpe DAC, Dixon K, Moghissi K. Endoscopic laser treatment for tracheal obstruction. Eur J Cardiothorac Surg 1996;10:722 6. [13] Gluth MB, Shinners PA, Kasperbauer JL. Subglottic stenosis associated with Wegener s granulomatosis. Laryngoscope 2003; 113:1304 7.

822 A.M. Ciccone et al. / European Journal of Cardio-thoracic Surgery 26 (2004) 818 822 [14] Giudice M, Piazza C, Foccoli P, Toninelli C, Cavaliere S, Peretti G. Idiopathic subglottic stenosis: management by endoscopic and openneck surgery in a series of 30 patients. Eur Arch Otorhinolaryngol 2003;260:235 8. [15] Shapshay SM, Beamis Jr JF, Hybels RL, Bohigian RK. Endoscopic treatment of subglottic and tracheal stenosis by radial laser incision and dilatation. Ann Otol Rhinol Laryngol 1987;96(6):661 4. [16] Grillo HC, Donahue DM, Mathisen DJ, Wain JC, Wright CD. Postintubation tracheal stenosis. Treatment and results. J Thorac Cardiovasc Surg 1995;109:486 93. [17] Bisson A, Bonnette P, El Kadi B. Tracheal sleeve resection for iatrogenic stenoses (subglottic laryngeal and tracheal). J Thorac Cardiovasc Surg 1992;104:882 7. Appendix A. Conference discussion Dr R. Santosham (India): We have had a fairly large experience of managing tracheal stenosis. We have done about 190 cases. But the thing you re talking about is laryngotracheal stenosis, and we understand that it s a difficult problem both surgically and with the use of laser. The only thing is that now the principles have been laid down and even higher strictures we are able to resect. The only question that I would like to ask is, what stent did you use in these cases? We have found the expandable stents very useless in these situations and most of them give rise to problems in benign tracheal disease. That has been our observation. Dr Rendina: We have used Dumon stents for a number of reasons. First of all, they can easily be removed at any time, they are reliable, they are cost-effective, and they do not cause the dreaded complication of granulation tissue on the upper and lower ends of the stent. As far as the laryngotracheal stenosis is concerned, maybe that would take too much time to comment, but the main difficulty is the anatomical location of the stenosis because the cricoid area is usually much more rigid than the rest of the trachea and the vicinity of the vocal cords makes a stent very unstable in that area. Dr Santosham: How are you able to maintain the stent in the position? The problem is that they tend to slip, especially in benign disease, in high tracheal stenosis. Our problem has been that it tends to slip down because it doesn t hold on. Dr Rendina: True. Dr T. Orlowski (Warsaw, Poland): So could you explain how you fix the stent and how you position it according to the vocal cord level? Dr Rendina: We place the stent with the normal stent deployer which has been designed and proposed by Dumon, and it s very easy to put the stent in. What is less easy and sometimes depends on the type of the stenosis, is to have the stent stay in place. Now, for laryngotracheal stenosis, there is a little trick. When you have a stenosis at the level of the upper rings of the trachea which makes the trachea rigid in that area, the rigidity of the trachea is comparable with the rigidity of the cricoid, and therefore the studs around the stent keep it in place. In other words, the more the stenosis is extended over the upper trachea, the more likely the stent is to stay in place. Dr G. Stamatis (Essen, Germany): Can you give us more information about the role of comorbidity in your decision as to which patients are suitable for operation and which ones for conservative treatment? Dr Rendina: Yes. I want to make this very clear. The nonoperative treatment in these patients is not an alternative to surgery. Sometimes the stenosis is fresh, with some degree of inflamation and edema and you do not want to operate; in these cases the alternative is to make a permanent tracheostomy, which we don t like to do for the risk of infection that it carries with it. Also, the patients in whom we have used nonoperative treatment were either very old or tetraplegic, because laryngo-tracheal problems usually occur in patients who have other comorbidities. The young patients in whom we have used this approach were patients with serious neurological problems or patients who were hospitalized and intubated for cardiopulmonary reasons. Dr I. Poliakov (Krasnodar, Russian Federation): Did you observe any granulation tissue growth below or in the upper margin of the stent resulting in extension of the stenosis after you removed the stent? Dr Rendina: We did not. Dr Poliakov: How many patients did you convert to surgery after conservative treatment failed? Dr Rendina: As I said, 4. Dr P. Macchiarini (Hannover, Germany): I have a comment about group 3 where 8 patients were operated only and you have a 25% stenosis. Do you have any technical reasons for that or indications or what? Dr Rendina: No. Two patients in the group you are referring to had restenosis after surgery. One was a 50-year-old patient with a congenital stenosis probably dating back to his early years of life, which was misconsidered for many years, and he had a restenosis which was treated by dilatation and stenting and healed after a year. Another patient had tracheostomy done elsewhere. He had some infection of the subcutaneous tissues and maybe that might have had an impact on restenosis.