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Surgical Treatment of Congenital Distal Tracheal Stenosis Involving the Carina
CASE REPORTS
Surgical Treatment of Congenital Distal Tracheal Stenosis Involving the Carina Naoki Minato, M.D., Kenji Itoh, M.D., Yasunori Ohkawa, M.D., Hitoshi Matsunaga, M.D., Nobuhiro Nagata, M.D., and Osamu Fukatsu, M.D. ABSTRACT Untreated congenital stenosis of the distal trachea frequently results in lethal airway obstruction.A 3year-old boy with segmental stenosis of the distal trachea and a 2-year-old girl with segmental stenosis involving the carina and the right main bronchus were treated successfully with resection and reanastomosis. Operative techniques, anesthetic management, postoperative care, and tracheal growth after anastomosis are discussed. Congenital tracheal stenosis causes congenital stridor and intractable pneumonia that occasionally can be lethal because of sudden obstruction of the stenotic trachea due to secretion or inflammatory edema. Medical therapy is not effective for this lesion, and surgical repair is needed. We describe 2 children with congenital tracheal stenosis treated successfully by resection and reanastomosis.
Patient 1 A 3-year-old boy had suffered from stridor since birth. As a newborn he had undergone operation for congenital duodenal atresia. He was admitted to our hospital because of repeated episodes of pneumonia. Chest roentgenogram demonstrated pneumonia of the right upper and lower lung fields. Bronchoscopy showed distal tracheal stenosis. A bronchogram revealed a tracheal bronchus and stenosis from just below the tracheal bronchus to the carina; the distal portion of the bronchus was normal (Fig 1). Through a right anterolateral thoracotomy at the third intercostal space and an upper median sternotomy, a 2.8-cm stenotic segment was resected from just below the tracheal bronchus to the bifurcation. The airway had an inner diameter of 1.5 mm. An end-to-end anastomosis then was performed with interrupted 4-0 Dexon sutures while the left lung was ventilated with an endotracheal tube inserted from the operative field. To minimize tension at the anastomosis, the patient’s chin was sutured to the presternal skin to keep his neck flexed for two weeks postoperatively. The patient was extubated on the second postopera-
From the Divisions of Thoracic and Cardiovascular Surgery and Anesthesiology, Kanagawa Children’s Medical Center, Yokohama, Japan. Accepted for publication Nov 14, 1985 Address reprint requests to Dr. Minato, Division of Thoracic and Cardiovascular Surgery, Saga Medical School, Sanbonsugi, Nabeshima, Saga, Japan.
326 Ann Thorac Surg 42:326-328, Sep 1986
tive day. Bronchoscopy revealed no stenosis and no granulation at the anastomosis. Air tracheogram demonstrated no stenosis of the carina. Stridor completely disappeared, and in the five years since operation, he has had no symptoms of restenosis, such as pneumonia or respiratory distress.
Patient 2 A 2-year-old girl was seen with congenital stridor and repeated episodes of pneumonia that necessitated frequent hospitalization. Chest roentgenogram showed bilateral pneumonia of the lower lung fields, and bronchogram demonstrated a tracheal bronchus and stenosis of the trachea from just below the tracheal bronchus to the bifurcation, the long segment of the right main bronchus, and the orifice of the left main bronchus (Fig 2). Right anterolateral thoracotomy and upper median sternotomy were performed as in Patient 1. A 1.4-cm segment of stenotic trachea was resected from just below the tracheal bronchus to the bifurcation. The airway had an inner diameter of 1 mm. Incisions of the tracheal bronchus and the right main bronchus were needed to enlarge the anastomotic stoma. The left lung was ventilated using high-frequency jet ventilation, and the tracheal bronchus was ventilated with a nasotracheal tube. End-to-end anastomosis was accomplished with interrupted 4-0 monofilament sutures while the patient’s head was maintained in a flexed position. The patient’s chin was sutured to the presternal skin to keep her neck flexed for two weeks postoperatively. Her postoperative course was complicated by tracheobronchial secretions and right middle and lower lobe atelectasis caused by a narrow right main bronchus. She was extubated 19 days postoperation. Bronchogram revealed obstruction of the right main bronchus but no stenosis at the anastomosis. Although the atelectasis of the right middle and lower lobes did not improve, the patient had neither fever nor leukocytosis. Seven months after the operation, she is doing well.
Comment Cantrell and Guild [l]have classified congenital tracheal stenosis into the following three groups: (1)generalized hypoplasia, (2) funnellike stenosis, and (3) segmental stenosis. Segmental stenosis is the most common anomaly, and it often has an abnormal origin in the major bronchi. In all groups, the membranous trachea is usually absent with fusion of the cartilages in the stenotic area. The survival rate following attempted tracheal resec-
327 Case Report: Minato, Itoh, Ohkawa, et al: Treating Congenital Distal Tracheal Stenosis
Fig I. (Patient 2 .) Preoperative bronchogram.
Fig 2. (Patient 2.) Preoperative bronchogram.
tion for congenital lesions in infants and children is low, especially in those in whom the carina and the main bronchus are involved [2-51. Tracheal resection and reanastomosis usually can be performed in funnellike and segmental stenosis, while reconstruction using autologous esophagus (61 and costal cartilage grafts [7] has been reported in generalized hypoplasia. Difficulties of anastomotic technique caused by the small size of the trachea and problems with intraoperative anesthetic management also lower the chances of successful operation. In our two patients, obstruction of stenotic trachea
by tracheobronchial secretions caused ventilatory failure during tracheal dissection and deterioration in blood gas values; Patient 1arterial oxygen tension (PaOz), 90 to 130 mm Hg; arterial carbon dioxide tension (PaC02), 65 to 120 mm Hg; pH, 7.0 to 7.2; Patient 2: PaOz, 70 to 220 mm Hg; PaC02, 65 to 115 mm Hg; pH, 6.9 to 7.2. During tracheal anastomosis, blood gas values remained rather poor regardless of whether one-lung ventilation through endotracheal tube (Patient 1: Pa02, 200-300 mm Hg; PaC02, 75-90 mm Hg; pH, 7.0-7.2) or high-frequency jet ventilation was used (Patient 2: PaO2,70-120 mm Hg; PaC02, 50-70 mm Hg; pH, 7.2-7.3); however, use of high-frequency jet ventilation seemed to maintain the patient’s PaC02 and pH more effectively (Fig 3).
328 The Annals of Thoracic Surgery Vol 42 No 3 September 1986
"
0' PCO?
Pcoz op
Completion of anastomosis
stenotic trachea
c
End
c
50 .
I 0'0
bit lung1
lrtNTlLATlON
50
100
150
0-
.
bilateral lung
200
25OM~n
I 0
HFJV
HFJV
/A
bilateral lung @'
50
c
Fig 3. lntraoperative changes in blood gas values in Patients 1 (lefthand column) and 2 (right-hand column). (POp= oxygen tension; PC02 = carbon dioxide tension; Op = operation; HFJV = highfrequency jet ventilation.)
To minimize tension at the anastomotic site, the anastomosis was performed with the patient in neck-flexed position; in addition, the chin was sutured to the presternal skin to maintain cervical flexion for two weeks postoperatively. Tracheal stricture after the anastomosis seems to depend on tension at the anastomotic site, inflammatory reaction, or suture material [8,9]. The endotracheal tube should be extubated as soon as possible because of the risk of leak, separation, or restenosis at the anastomosis. No serious complications occurred in Patient 2 despite the long intubation. In young patients there is the long-term problem of growth of the anastomosis. Maeda and Grillo [lo] have shown growth of the anastomosis in puppies. Patient 1 has shown no symptoms of restenosis, such as stridor or pneumonia, and an air tracheogram revealed no stenosis of the anastomosis five years after the operation. There seems to be adequate anastomotic growth clinically. The long segmental stenosis of the right main bronchus in Patient 2 seemed to present two alternatives for operation, lobectomy of the right middle and lower lobes, or no lobectomy. We selected the latter approach with an expectation that the right middle and lower lobes could be kept expanded. Unfortunately, atelectasis occurred in that portion; thus a right middle and lower lobectomy will be necessary if she suffers from recurrent pneumonia. Careful followup is needed to establish the adequacy of the operation.
3
bilateral lung
5
I 0 Min
In conclusion, the keys to performing successful tracheal reconstruction in infants and children are (1) keeping good ventilatory conditions during operation (we prefer the use of high-frequency jet ventilation during anastomosis) and (2) keeping the patient's neck flexed during anastomosis and postoperatively for two or more weeks to decrease tension at the anastomosis.
References 1. Cantrell JR, Guild HG: Congenital stenosis of the trachea.
Am J Surg 108:297, 1964 2. Mansfield PB: Tracheal resection in infancy. J Pediatr Surg
15:79, 1980 3. Harrison MR, Heldt GP, Brasch RC, et al: Resection of distal tracheal stenosis in a baby with agenesis of the lung. J Pediatr Surg 15:938, 1980 4. Weber TR, Eisen H, Scott PH, et al: Resection of congenital tracheal stenosis involving the canna. J Thorac Cardiovasc Surg 84:200, 1982 5. Nakayama DK, Harrison MR, Lorimier AA, et al: Reconstructive surgery for obstructing lesions of the intrathoracic trachea in infants and small children. J Pediatr Surg 17854, 1982 6. Fonkalsrud EW, Sumida S: Tracheal replacement with autologous esophagus for tracheal stricture. Arch Surg 102:139, 1971 7. Kimura K: A new technique for repair of congenital tracheal stenosis involving the entire trachea. Jpn J Pediatr Surg 14:1267, 1982 8. Maeda M, Grillo HC: Effect of tension on tracheal growth after resection and anastomosis in puppies. J Thorac Cardiovasc Surp. 65:658, 1973 9. Kotake Y, G>llo HC: Reduction of tension at the anastomosis following tracheal resection in puppies. J Thorac Cardiovasc Surg 71:603, 1976 10. Maeda M, Grillo HC: Tracheal growth following anastomosis in puppies. J Thorac Cardiovasc Surg 64:304, 1972
Surgical Treatment of Congenital Distal Tracheal Stenosis Involving the Carina Naoki Minato, M.D., Kenji Itoh, M.D., Yasunori Ohkawa, M.D., Hitoshi Matsunaga, M.D., Nobuhiro Nagata, M.D., and Osamu Fukatsu, M.D. ABSTRACT Untreated congenital stenosis of the distal trachea frequently results in lethal airway obstruction.A 3year-old boy with segmental stenosis of the distal trachea and a 2-year-old girl with segmental stenosis involving the carina and the right main bronchus were treated successfully with resection and reanastomosis. Operative techniques, anesthetic management, postoperative care, and tracheal growth after anastomosis are discussed. Congenital tracheal stenosis causes congenital stridor and intractable pneumonia that occasionally can be lethal because of sudden obstruction of the stenotic trachea due to secretion or inflammatory edema. Medical therapy is not effective for this lesion, and surgical repair is needed. We describe 2 children with congenital tracheal stenosis treated successfully by resection and reanastomosis.
Patient 1 A 3-year-old boy had suffered from stridor since birth. As a newborn he had undergone operation for congenital duodenal atresia. He was admitted to our hospital because of repeated episodes of pneumonia. Chest roentgenogram demonstrated pneumonia of the right upper and lower lung fields. Bronchoscopy showed distal tracheal stenosis. A bronchogram revealed a tracheal bronchus and stenosis from just below the tracheal bronchus to the carina; the distal portion of the bronchus was normal (Fig 1). Through a right anterolateral thoracotomy at the third intercostal space and an upper median sternotomy, a 2.8-cm stenotic segment was resected from just below the tracheal bronchus to the bifurcation. The airway had an inner diameter of 1.5 mm. An end-to-end anastomosis then was performed with interrupted 4-0 Dexon sutures while the left lung was ventilated with an endotracheal tube inserted from the operative field. To minimize tension at the anastomosis, the patient’s chin was sutured to the presternal skin to keep his neck flexed for two weeks postoperatively. The patient was extubated on the second postopera-
From the Divisions of Thoracic and Cardiovascular Surgery and Anesthesiology, Kanagawa Children’s Medical Center, Yokohama, Japan. Accepted for publication Nov 14, 1985 Address reprint requests to Dr. Minato, Division of Thoracic and Cardiovascular Surgery, Saga Medical School, Sanbonsugi, Nabeshima, Saga, Japan.
326 Ann Thorac Surg 42:326-328, Sep 1986
tive day. Bronchoscopy revealed no stenosis and no granulation at the anastomosis. Air tracheogram demonstrated no stenosis of the carina. Stridor completely disappeared, and in the five years since operation, he has had no symptoms of restenosis, such as pneumonia or respiratory distress.
Patient 2 A 2-year-old girl was seen with congenital stridor and repeated episodes of pneumonia that necessitated frequent hospitalization. Chest roentgenogram showed bilateral pneumonia of the lower lung fields, and bronchogram demonstrated a tracheal bronchus and stenosis of the trachea from just below the tracheal bronchus to the bifurcation, the long segment of the right main bronchus, and the orifice of the left main bronchus (Fig 2). Right anterolateral thoracotomy and upper median sternotomy were performed as in Patient 1. A 1.4-cm segment of stenotic trachea was resected from just below the tracheal bronchus to the bifurcation. The airway had an inner diameter of 1 mm. Incisions of the tracheal bronchus and the right main bronchus were needed to enlarge the anastomotic stoma. The left lung was ventilated using high-frequency jet ventilation, and the tracheal bronchus was ventilated with a nasotracheal tube. End-to-end anastomosis was accomplished with interrupted 4-0 monofilament sutures while the patient’s head was maintained in a flexed position. The patient’s chin was sutured to the presternal skin to keep her neck flexed for two weeks postoperatively. Her postoperative course was complicated by tracheobronchial secretions and right middle and lower lobe atelectasis caused by a narrow right main bronchus. She was extubated 19 days postoperation. Bronchogram revealed obstruction of the right main bronchus but no stenosis at the anastomosis. Although the atelectasis of the right middle and lower lobes did not improve, the patient had neither fever nor leukocytosis. Seven months after the operation, she is doing well.
Comment Cantrell and Guild [l]have classified congenital tracheal stenosis into the following three groups: (1)generalized hypoplasia, (2) funnellike stenosis, and (3) segmental stenosis. Segmental stenosis is the most common anomaly, and it often has an abnormal origin in the major bronchi. In all groups, the membranous trachea is usually absent with fusion of the cartilages in the stenotic area. The survival rate following attempted tracheal resec-
327 Case Report: Minato, Itoh, Ohkawa, et al: Treating Congenital Distal Tracheal Stenosis
Fig I. (Patient 2 .) Preoperative bronchogram.
Fig 2. (Patient 2.) Preoperative bronchogram.
tion for congenital lesions in infants and children is low, especially in those in whom the carina and the main bronchus are involved [2-51. Tracheal resection and reanastomosis usually can be performed in funnellike and segmental stenosis, while reconstruction using autologous esophagus (61 and costal cartilage grafts [7] has been reported in generalized hypoplasia. Difficulties of anastomotic technique caused by the small size of the trachea and problems with intraoperative anesthetic management also lower the chances of successful operation. In our two patients, obstruction of stenotic trachea
by tracheobronchial secretions caused ventilatory failure during tracheal dissection and deterioration in blood gas values; Patient 1arterial oxygen tension (PaOz), 90 to 130 mm Hg; arterial carbon dioxide tension (PaC02), 65 to 120 mm Hg; pH, 7.0 to 7.2; Patient 2: PaOz, 70 to 220 mm Hg; PaC02, 65 to 115 mm Hg; pH, 6.9 to 7.2. During tracheal anastomosis, blood gas values remained rather poor regardless of whether one-lung ventilation through endotracheal tube (Patient 1: Pa02, 200-300 mm Hg; PaC02, 75-90 mm Hg; pH, 7.0-7.2) or high-frequency jet ventilation was used (Patient 2: PaO2,70-120 mm Hg; PaC02, 50-70 mm Hg; pH, 7.2-7.3); however, use of high-frequency jet ventilation seemed to maintain the patient’s PaC02 and pH more effectively (Fig 3).
328 The Annals of Thoracic Surgery Vol 42 No 3 September 1986
"
0' PCO?
Pcoz op
Completion of anastomosis
stenotic trachea
c
End
c
50 .
I 0'0
bit lung1
lrtNTlLATlON
50
100
150
0-
.
bilateral lung
200
25OM~n
I 0
HFJV
HFJV
/A
bilateral lung @'
50
c
Fig 3. lntraoperative changes in blood gas values in Patients 1 (lefthand column) and 2 (right-hand column). (POp= oxygen tension; PC02 = carbon dioxide tension; Op = operation; HFJV = highfrequency jet ventilation.)
To minimize tension at the anastomotic site, the anastomosis was performed with the patient in neck-flexed position; in addition, the chin was sutured to the presternal skin to maintain cervical flexion for two weeks postoperatively. Tracheal stricture after the anastomosis seems to depend on tension at the anastomotic site, inflammatory reaction, or suture material [8,9]. The endotracheal tube should be extubated as soon as possible because of the risk of leak, separation, or restenosis at the anastomosis. No serious complications occurred in Patient 2 despite the long intubation. In young patients there is the long-term problem of growth of the anastomosis. Maeda and Grillo [lo] have shown growth of the anastomosis in puppies. Patient 1 has shown no symptoms of restenosis, such as stridor or pneumonia, and an air tracheogram revealed no stenosis of the anastomosis five years after the operation. There seems to be adequate anastomotic growth clinically. The long segmental stenosis of the right main bronchus in Patient 2 seemed to present two alternatives for operation, lobectomy of the right middle and lower lobes, or no lobectomy. We selected the latter approach with an expectation that the right middle and lower lobes could be kept expanded. Unfortunately, atelectasis occurred in that portion; thus a right middle and lower lobectomy will be necessary if she suffers from recurrent pneumonia. Careful followup is needed to establish the adequacy of the operation.
3
bilateral lung
5
I 0 Min
In conclusion, the keys to performing successful tracheal reconstruction in infants and children are (1) keeping good ventilatory conditions during operation (we prefer the use of high-frequency jet ventilation during anastomosis) and (2) keeping the patient's neck flexed during anastomosis and postoperatively for two or more weeks to decrease tension at the anastomosis.
References 1. Cantrell JR, Guild HG: Congenital stenosis of the trachea.
Am J Surg 108:297, 1964 2. Mansfield PB: Tracheal resection in infancy. J Pediatr Surg
15:79, 1980 3. Harrison MR, Heldt GP, Brasch RC, et al: Resection of distal tracheal stenosis in a baby with agenesis of the lung. J Pediatr Surg 15:938, 1980 4. Weber TR, Eisen H, Scott PH, et al: Resection of congenital tracheal stenosis involving the canna. J Thorac Cardiovasc Surg 84:200, 1982 5. Nakayama DK, Harrison MR, Lorimier AA, et al: Reconstructive surgery for obstructing lesions of the intrathoracic trachea in infants and small children. J Pediatr Surg 17854, 1982 6. Fonkalsrud EW, Sumida S: Tracheal replacement with autologous esophagus for tracheal stricture. Arch Surg 102:139, 1971 7. Kimura K: A new technique for repair of congenital tracheal stenosis involving the entire trachea. Jpn J Pediatr Surg 14:1267, 1982 8. Maeda M, Grillo HC: Effect of tension on tracheal growth after resection and anastomosis in puppies. J Thorac Cardiovasc Surp. 65:658, 1973 9. Kotake Y, G>llo HC: Reduction of tension at the anastomosis following tracheal resection in puppies. J Thorac Cardiovasc Surg 71:603, 1976 10. Maeda M, Grillo HC: Tracheal growth following anastomosis in puppies. J Thorac Cardiovasc Surg 64:304, 1972