2
References 1. Rahman NA, Fruchter O, Shitrit D, Fox BD, Kramer MR. Flexible bronchoscopic management of benign tracheal ste- nosis: long term follow-up of 115 patients. J Cardiothorac Surg 2010;5:2. 2. Damrose EJ. On the development of idiopathic subglottic stenosis. Med Hypotheses 2008;71:122–5. 3. Couraud L, Jougon JB, Velly JF. Surgical treatment of non- tumoral stenoses of the upper airway. Ann Thorac Surg 1995;60:250 –9. 4. Ashiku SK, Kuzucu A, Grillo HC, et al. Idiopathic laryngo- tracheal stenosis: effective definitive treatment with laryngo- tracheal resection. J Thorac Cardiovasc Surg 2004;127:99 – 107. 5. Grillo HC, Mark EJ, Mathisen DJ, Wain JC. Idiopathic laryngotracheal stenosis and its management. Ann Thorac Surg 1993;56:80 –7. 6. Park SS, Streitz JM Jr, Rebeiz EE, Shapshay SM. Idiopathic subglottic stenosis. Arch Otolaryngol Head Neck Surg 1995; 121:894 –7. 7. Dedo HH, Catten MD. Idiopathic progressive subglottic stenosis: findings and treatment in 52 patients. Ann Otol Rhinol Laryngol 2001;110:305–11. 8. Rossi C, Colombari F, Guembarowsky AL, Ferreira Filho OF, Thomson JC. Idiopathic tracheal stenosis. A report of four cases. J Bras Pneumol 2007;33:101– 4. 9. Benjamin B, Jacobson I, Eckstein R. Idiopathic subglottic stenosis: diagnosis and endoscopic laser treatment. Ann Otol Rhinol Laryngol 1997;106:770 – 4. 10. Hans S, de Mones E, Biacabe B, et al. Idiopathic sub-glottal stenosis in the adult. Article in French Ann Otolaryngol Chir Cervicofac 1999;116:250 – 6. 11. Mayse ML, Greenheck J, Friedman M, Kovitz KL. Successful bronchoscopic balloon dilation of nonmalignant tracheo- bronchial obstruction without fluoroscopy. Chest 2004;126: 634 –7. 12. Freitag L, Ernst A, Unger M, Kovitz K, Marquette CH. A proposed classification system of central airway stenosis. Eur Respir J 2007;30:7–12. 13. Liberman M, Mathisen DJ. Treatment of idiopathic laryngo- tracheal stenosis. Semin Thorac Cardiovasc Surg 2009;21: 278 – 83. 14. Brichet A, Verkindre C, Dupont J, et al. Multidisciplinary approach to management of postintubation tracheal steno- ses. Eur Respir J 1999;13:888 –93. 15. Galluccio G, Lucantoni G, Battistoni P, et al. Interventional endoscopy in the management of benign tracheal stenoses: definitive treatment at long-term follow-up. Eur J Cardio- thorac Surg 2009;35:429 –33. 16. Mark EJ, Meng F, Kradin RL, Mathisen DJ, Matsubara O. Idiopathic tracheal stenosis: a clinicopathologic study of 63 cases and comparison of the pathology with chondromala- cia. Am J Surg Pathol 2008;32:1138 – 43. 17. Smith ME, Elstad M. Mitomycin C and the endoscopic treatment of laryngotracheal stenosis: are two applications better than one? Laryngoscope 2009;119:272– 83. 18. Toohill RJ, Ulualp SO, Shaker R. Evaluation of gastroesoph- ageal reflux in patients with laryngotracheal stenosis. Ann Otol Rhinol Laryngol 1998;107:1010 – 4. 19. Terra RM, de Medeiros IL, Minamoto H, Nasi A, Pego- Fernandes PM, Jatene FB. Idiopathic tracheal stenosis: suc- cessful outcome with antigastroesophageal reflux disease therapy. Ann Thorac Surg 2008;85:1438 –9. 20. Valdez TA, Shapshay SM. Idiopathic subglottic stenosis revisited. Ann Otol Rhinol Laryngol 2002;111:690 –5. INVITED COMMENTARY Idiopathic tracheal stenosis (ITS) is a rare disease char- acterized by inflammation and stenosis of the proximal airway. Inflammation is most severe at the level of the cricoid cartilage, although the proximal trachea or sub- glottis may be involved. Interestingly, the disease is almost always confined to young women. Physicians caring for these patients have several chal- lenges. The first is to provide an accurate diagnosis. ITS is a diagnosis of exclusion and other diseases, particu- larly collagen vascular disorders such as Wegener’s gran- ulomatosis, should be ruled out. The distinction between ITS and these other conditions is more than academic. Airway involvement with collagen vascular disease may respond promptly to corticosteroids and other immuno- suppressive medication. Furthermore, the inflammatory lesions seen with collagen vascular diseases are migra- tory. Operating on such a patient may lead to recurrent inflammation at the anastomosis and a failure of surgical therapy. In contrast, the lesions of ITS are thought to be stable and unlikely to recur after operation. Resection and reconstruction of the airway should therefore lead to a durable benefit in the majority of patients. This is indeed the result in the largest surgical series of these patients [1]. Of 73 patients operated on for ITS, there were no deaths and no reoperations. With 8 years of follow-up, results were good to excellent in 90% of patients. In contrast to the Massachusetts General Hospital (Boston, MA) series, Perotin and colleagues [2] reported the results of endoscopic treatment of ITS among several centers in France. Endoscopic treatment included balloon dilation, laser or electrocoagulation, and stent insertion. As one would expect, endoscopic management of ITS was safe and effective in improving symptoms of dyspnea and stridor. However the recurrence rate was high (87% at 5 years) and most patients required repeated interventions. The high recurrence rate in the present series should not allow one to conclude that laryngotracheal resection should be the standard of care for all patients with ITS. Endoscopic therapy is not only safe and effective in the short term but can also be performed in many centers. In contrast, laryngotracheal resection is an exceptionally demanding procedure, and the excellent results from Ashiku and colleagues [1] reflect their considerable ex- perience. It should not be assumed that the results from centers with less experience would be equivalent. The other issue is the selection of patients and the timing of the surgical procedure. Patients with lesions that extend to the vocal cords should not undergo oper- ation. Symptoms can be palliated with endoscopic treat- ment, and operation can be considered at a later date. The experience of Perotin and colleagues [2] would indicate that some of these patients may have a durable response to dilation, and surgical procedures can be avoided. 301 Ann Thorac Surg PEROTIN ET AL 2011;92:297–302 ENDOSCOPIC MANAGEMENT OF ITS © 2011 by The Society of Thoracic Surgeons 0003-4975/$36.00 Published by Elsevier Inc doi:10.1016/j.athoracsur.2011.04.080 GENERAL THORACIC

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Page 1: Invited Commentary

301Ann Thorac Surg PEROTIN ET AL2011;92:297–302 ENDOSCOPIC MANAGEMENT OF ITS

GEN

ERA

LT

HO

RA

CIC

References

1. Rahman NA, Fruchter O, Shitrit D, Fox BD, Kramer MR.Flexible bronchoscopic management of benign tracheal ste-nosis: long term follow-up of 115 patients. J CardiothoracSurg 2010;5:2.

2. Damrose EJ. On the development of idiopathic subglotticstenosis. Med Hypotheses 2008;71:122–5.

3. Couraud L, Jougon JB, Velly JF. Surgical treatment of non-tumoral stenoses of the upper airway. Ann Thorac Surg1995;60:250–9.

4. Ashiku SK, Kuzucu A, Grillo HC, et al. Idiopathic laryngo-tracheal stenosis: effective definitive treatment with laryngo-tracheal resection. J Thorac Cardiovasc Surg 2004;127:99–107.

5. Grillo HC, Mark EJ, Mathisen DJ, Wain JC. Idiopathiclaryngotracheal stenosis and its management. Ann ThoracSurg 1993;56:80–7.

6. Park SS, Streitz JM Jr, Rebeiz EE, Shapshay SM. Idiopathicsubglottic stenosis. Arch Otolaryngol Head Neck Surg 1995;121:894–7.

7. Dedo HH, Catten MD. Idiopathic progressive subglotticstenosis: findings and treatment in 52 patients. Ann OtolRhinol Laryngol 2001;110:305–11.

8. Rossi C, Colombari F, Guembarowsky AL, Ferreira Filho OF,Thomson JC. Idiopathic tracheal stenosis. A report of fourcases. J Bras Pneumol 2007;33:101–4.

9. Benjamin B, Jacobson I, Eckstein R. Idiopathic subglotticstenosis: diagnosis and endoscopic laser treatment. AnnOtol Rhinol Laryngol 1997;106:770–4.

10. Hans S, de Mones E, Biacabe B, et al. Idiopathic sub-glottal

stenosis in the adult. Article in French Ann Otolaryngol ChirCervicofac 1999;116:250–6.

In contrast to the Massachusetts General Hospital

© 2011 by The Society of Thoracic SurgeonsPublished by Elsevier Inc

11. Mayse ML, Greenheck J, Friedman M, Kovitz KL. Successfulbronchoscopic balloon dilation of nonmalignant tracheo-bronchial obstruction without fluoroscopy. Chest 2004;126:634–7.

12. Freitag L, Ernst A, Unger M, Kovitz K, Marquette CH. Aproposed classification system of central airway stenosis.Eur Respir J 2007;30:7–12.

13. Liberman M, Mathisen DJ. Treatment of idiopathic laryngo-tracheal stenosis. Semin Thorac Cardiovasc Surg 2009;21:278–83.

14. Brichet A, Verkindre C, Dupont J, et al. Multidisciplinaryapproach to management of postintubation tracheal steno-ses. Eur Respir J 1999;13:888–93.

15. Galluccio G, Lucantoni G, Battistoni P, et al. Interventionalendoscopy in the management of benign tracheal stenoses:definitive treatment at long-term follow-up. Eur J Cardio-thorac Surg 2009;35:429–33.

16. Mark EJ, Meng F, Kradin RL, Mathisen DJ, Matsubara O.Idiopathic tracheal stenosis: a clinicopathologic study of 63cases and comparison of the pathology with chondromala-cia. Am J Surg Pathol 2008;32:1138–43.

17. Smith ME, Elstad M. Mitomycin C and the endoscopictreatment of laryngotracheal stenosis: are two applicationsbetter than one? Laryngoscope 2009;119:272–83.

18. Toohill RJ, Ulualp SO, Shaker R. Evaluation of gastroesoph-ageal reflux in patients with laryngotracheal stenosis. AnnOtol Rhinol Laryngol 1998;107:1010–4.

19. Terra RM, de Medeiros IL, Minamoto H, Nasi A, Pego-Fernandes PM, Jatene FB. Idiopathic tracheal stenosis: suc-cessful outcome with antigastroesophageal reflux diseasetherapy. Ann Thorac Surg 2008;85:1438–9.

20. Valdez TA, Shapshay SM. Idiopathic subglottic stenosisrevisited. Ann Otol Rhinol Laryngol 2002;111:690–5.

INVITED COMMENTARY

Idiopathic tracheal stenosis (ITS) is a rare disease char-acterized by inflammation and stenosis of the proximalairway. Inflammation is most severe at the level of thecricoid cartilage, although the proximal trachea or sub-glottis may be involved. Interestingly, the disease isalmost always confined to young women.

Physicians caring for these patients have several chal-lenges. The first is to provide an accurate diagnosis. ITSis a diagnosis of exclusion and other diseases, particu-larly collagen vascular disorders such as Wegener’s gran-ulomatosis, should be ruled out. The distinction betweenITS and these other conditions is more than academic.Airway involvement with collagen vascular disease mayrespond promptly to corticosteroids and other immuno-suppressive medication. Furthermore, the inflammatorylesions seen with collagen vascular diseases are migra-tory. Operating on such a patient may lead to recurrentinflammation at the anastomosis and a failure of surgicaltherapy.

In contrast, the lesions of ITS are thought to be stableand unlikely to recur after operation. Resection andreconstruction of the airway should therefore lead to adurable benefit in the majority of patients. This is indeedthe result in the largest surgical series of these patients[1]. Of 73 patients operated on for ITS, there were nodeaths and no reoperations. With 8 years of follow-up,results were good to excellent in 90% of patients.

(Boston, MA) series, Perotin and colleagues [2] reportedthe results of endoscopic treatment of ITS among severalcenters in France. Endoscopic treatment included balloondilation, laser or electrocoagulation, and stent insertion.As one would expect, endoscopic management of ITSwas safe and effective in improving symptoms of dyspneaand stridor. However the recurrence rate was high (87%at 5 years) and most patients required repeatedinterventions.

The high recurrence rate in the present series shouldnot allow one to conclude that laryngotracheal resectionshould be the standard of care for all patients with ITS.Endoscopic therapy is not only safe and effective in theshort term but can also be performed in many centers. Incontrast, laryngotracheal resection is an exceptionallydemanding procedure, and the excellent results fromAshiku and colleagues [1] reflect their considerable ex-perience. It should not be assumed that the results fromcenters with less experience would be equivalent.

The other issue is the selection of patients and thetiming of the surgical procedure. Patients with lesionsthat extend to the vocal cords should not undergo oper-ation. Symptoms can be palliated with endoscopic treat-ment, and operation can be considered at a later date.The experience of Perotin and colleagues [2] wouldindicate that some of these patients may have a durableresponse to dilation, and surgical procedures can be

avoided.

0003-4975/$36.00doi:10.1016/j.athoracsur.2011.04.080

Page 2: Invited Commentary

302 PEROTIN ET AL Ann Thorac SurgENDOSCOPIC MANAGEMENT OF ITS 2011;92:297–302

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Michael Kent, MD

Department of SurgeryBeth Israel Deaconess Medical Center110 Francis StSte 2ABoston, MA 02215

e-mail: [email protected]

References

1. Ashiku S, Kuzucu A, Grillo H, et al. Idiopathic laryngotra-cheal stenosis: effective definitive treatment with larnygotra-cheal resection. J Thorac Cardiovasc Surg 2002;127:99–107.

2. Perotin J-M, Jeanfaivre T, Thibout Y, et al. Endoscopic man-agement of idiopathic tracheal stenosis. Ann Thorac Surg

2011;92:297–302.