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Asian Cardiovasc Thorac Ann 2004;12:190-192
© 2004 Asia Publishing EXchange Ltd


ORIGINAL CONTRIBUTION

Slide Tracheoplasty for Congenital Tracheal Stenosis with Glottic Stenosis

Mohammed A Rafay, FRCS, Waseem M Hajjar, FRCS, Mohammed A Essa, MD, Ziyad Z Mahasin, FRCS, Khaled M Al-Kattan, FRCS

Department of Surgery, King Faisal Specialist Hospital and Research, Center Riyadh, Saudi Arabia

For reprint information contact: Khaled M Al-Kattan, FRCS Tel: 966 1 467 1920 Fax: 966 1 467 9494 Email: alkattan{at}ksu.edu.sa Thoracic Surgery Division, Department of Surgery (37), King Khalid University Hospital, College of Medicine, P.O. Box 7805, Riyadh 11472, Saudi Arabia.


    ABSTRACT
 TOP
 ABSTRACT
 INTRODUCTION
 DISCUSSION
 REFERENCES
 
A 7-month-old child with respiratory distress was diagnosed to have full-length tracheal stenosis with glottic stenosis. Slide tracheoplasty and anterior cricoid split with stenting for glottic stenosis were successfully performed, and the child has completed 5 years of asymptomatic follow-up. This case widens the scope for slide tracheoplasty, and cricoid split with stenting is a feasible procedure for associated glottic stenosis.


    INTRODUCTION
 TOP
 ABSTRACT
 INTRODUCTION
 DISCUSSION
 REFERENCES
 
Slide tracheoplasty repair for long segment congenital funnel-shaped trachea was first introduced in 1989 and its role in the management of congenital funnel-shaped trachea is well established. We report a case of congenital full-length tracheal stenosis with glottic stenosis. Slide tracheoplasty and anterior cricoid split with stenting for glottic stenosis successfully corrected the deformity. In addition, the patient has remained asymptomatic for 5 years since.

A 7-month-old male Ethiopian child with respiratory distress was referred to our thoracic surgical unit in July 1997. The offspring of first-cousin parents, his was a full-term normal vaginal delivery with birth weight of 3.5 kg. At birth, he had severe shortness of breath, wheezy chest and bilateral coarse crepitations. A diagnosis of meconium aspiration was made requiring neonatal intensive care, and he developed frequent episodes of respiratory distress over a 50-day intensive-care stay. Although he was diagnosed to have a congenital disorder of pyroglutamic acidemia, the episodes of respiratory distress persisted after the acidemia was corrected. His parents had lost one sibling at the age of 2 years at another hospital with a similar problem.

Until the age of 7 months, he frequently developed respiratory distress requiring hospital admissions. Wheezing, biphasic stridor and cyanosis lead to bronchoscopic examination. Stenosis of the airway starting from the cricoid down to the carina, allowing only a size 2.5-mm telescope to pass, was noted and confirmed by a computed tomography (CT) scan of the chest (Figure 1Go). The trachea had complete cartilaginous rings, and the right and left main bronchi were normal. Cardiac and systemic examination did not reveal any anomaly.



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Figure 1. Preoperative CT scan of the chest showing narrowed trachea.

 
Slide tracheoplasty with cricoid split was performed with a 2.5-mm internal diameter endotracheal tube. Through a collar incision, the cricoid and the entire trachea were exposed anteriorly showing cylindrical narrowing down to the carina. The trachea was transected horizontally in the mid way, the cephaloid stump was divided in the midline posteriorly and the caudad stump was divided anteriorly up to the carina. Slide tracheoplasty was performed by flexing the neck and sliding the cephaloid stump over the caudad stump. The anastomosis was constructed with dexon 5.0 interrupted sutures. The cricoid was split anteriorly and stented with a 4.5-mm internal diameter endotracheal tube (Figure 2Go).





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Figure 2. A diagram of the operative procedure showing: (A) the trachea divided horizontally in the mid way, the distal segment opened anteriorly and the proximal segment opened posteriorly; (B) the two segments brought together; and (C) complete anastomosis performed with cricoid split.

 
Bronchoscopic assessment under general anesthesia on the 8th postoperative day showed a satisfactory tracheal lumen and healing of the anastomosis. He was extubated, and further recovery was uneventful, with discharge home on the 10th postoperative day. To date, he has completed 5 years of asymptomatic follow-up with normal developmental milestones. Chest radiography (Figure 3Go) and a CT scan of the chest (Figure 4Go) showed normal tracheal diameter.



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Figure 3. Postoperative chest radiograph in posteroanterior view showing normal tracheal air column.

 


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Figure 4. Postoperative CT scan of the chest showing normal diameter of the trachea.

 

    DISCUSSION
 TOP
 ABSTRACT
 INTRODUCTION
 DISCUSSION
 REFERENCES
 
Congenital full-length tracheal stenosis (CTS) is a rare life-threatening anomaly characterized by the absence of the membranous portion, with fusion of each individual tracheal cartilage posteriorly, throughout the length of the trachea. Cantrell and Guild1 described different categories of long-segment congenital tracheal stenosis as generalized hypoplasia, funnel-shaped stenosis and segmental stenosis. In our case, the patient had been symptomatic from birth and was diagnosed with full-length CTS at 7 months. This diagnosis is often delayed from 2 days to 5 years depending upon the severity of stenosis.2

Bronchoscopy is diagnostic and can assess the extent and type of anomaly, as well as the status of both the upper airway and the distal bronchial tree. Patients with long-segment CTS are known to have associated pulmonary, cardiovascular, gastrointestinal and urogenital tract congenital anomalies, and appropriate investigations should be carried out to detect these. However, our patient had CTS with glottic stenosis and congenital pyroglutamic acidemia; associations not earlier reported. Slide tracheoplasty for full-length CTS was performed as described by Grillo in 1994,3 which was a modification of the procedure first performed by Goldstraw in 1989 and reported by Tsang et al.4 Generalized hypoplastic CTS was considered an untreatable condition and various procedures using autologous, homologous and heterologous patch graft or "gusset" procedures have been described.4,5,6 It was observed that gusset procedures required postoperative intubation for stenting for up to 3 months and granulation tissue at the site of patch graft often lead to restenosis.

The maximum lengths of CTS repaired by slide tracheoplasty were 83% of tracheal length repaired by Grillo3 in 1994 in a 19-year-old female patient and 100% repaired by Dayan et al5 in 1997. Our patient had full-length CTS with glottic stenosis.

We performed anterior cricoid split and stenting with a 4.5-mm internal diameter endotracheal tube for glottic stenosis. Bronchoscopic assessment carried out on the 8th postoperative day revealed a satisfactory lumen and well-healed anastomosis. The patient has now had 5 years of follow-up with normal developmental milestones and enjoys a symptom-free, normal life. There is no evidence of growth-related restenosis at the anastomotic site or inadequate tracheal growth. Grillo et al7 have documented normal tracheal growth in the long-term follow-up of patients undergoing slide tracheoplasty, and in 1997, Macchiriani et al8 reported similar results in an animal study.

Slide tracheoplasty, originally designed for long-segment congenital funnel-shaped tracheal stenosis, is a feasible procedure for full-length CTS, while anterior cricoid split with stenting is a feasible procedure for associated glottic stenosis. An evaluation for any associated congenital anomaly should also be performed.


    REFERENCES
 TOP
 ABSTRACT
 INTRODUCTION
 DISCUSSION
 REFERENCES
 

  1. Cantrell JR, Guild HG. Congenital stenosis of the trachea. Am J Surg 1964;108:297–305.[Medline]

  2. Benjamim B, Pitkin J, Cohen D. Congenital tracheal stenosis. Ann Otol Rhinol Laryngol 1981;90:364–71.[Medline]

  3. Grillo HC. Slide tracheoplasty for long-segment congenital tracheal stenosis. Ann Thorac Surg 1994;58:613–21.[Abstract]

  4. Tsang V, Murday A, Gillbe C, Goldstraw P. Slide tracheoplasty for congenital funnel-shaped tracheal stenosis. Ann Thorac Surg 1989;48:632–5.[Abstract]

  5. Dayan SH, Dunham ME, Baker CL, Mavroudis C, Holinger LD. Slide tracheoplasty in the management of congenital tracheal stenosis. Ann Otol Rhinol Laryngol 1997;106:914–9.[Medline]

  6. Idriss FS, DeLeon SY, Ilbawi MN, Gerson CR, Tucker GF, Holinger L. Tracheoplasty with pericardial patch for extensive tracheal stenosis in infants and children. J Thorac Cardiovasc Surg 1984;88:527–36.[Abstract]

  7. Grillo HC, Wright CD, Vlahakes GJ, MacGillivray TE. Management of congenital tracheal stenosis by means of slide tracheoplasty or resection and reconstruction, with long term follow-up of growth after slide tracheoplasty. J Thorac Cardiovasc Surg 2002;123:145–52.[Abstract/Free Full Text]

  8. Macchiriani P, Dulmet E, de Montpreville V, Mazmanian GM, Chapelier A, Dartevelle P. Tracheal growth after slide tracheoplasty. J Thorac Cardiovasc Surg 1997;113:558–66.[Abstract/Free Full Text]





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