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Bronchoplasty for bronchial stenosis in a neonate: a case report
Bronchoplasty for Bronchial Stenosis in a Neonate: A Case Report By K. Prabhakaran and J.Z. Patankar Singapore
Bronchial stenosis is an uncommon problem in children. Management can be difficult because of the small luminal diameter and proximity of the lung parenchyma to the bronchial lesion. Bronchoplasty procedures have obvious advantages in children because of their long life expectancy. Longterm functional results are superior compared with pneumonectomy because of preserved lung tissue. A premature neonate weighing only 779 g at birth was born with severe respiratory distress syndrome complicated with hyperbillirubinemia and bronchopulmonary dysplasia. The child underwent bronchoplasty using a costal cartilage graft for a right main bronchus stenosis after 2 failed attempts at bronchoscopic dilatations. In spite of all complications, the
child improved sufficiently to be discharged without any oxygen dependency. Our case highlights the successful application of bronchoplasty in a very small baby. Preoperative and intraoperative bronchoscopy was helpful in localization of the endobronchial lesion. Understanding the principle, precise attention to technical detail and meticulous postoperative care contribute toward long-term benefit for pulmonary conservation. J Pediatr Surg 39:E6-E8. © 2004 Elsevier Inc. All rights reserved.
B
The baby had a bronchopleural fistula. In view of failure of all conservative measures and clinical deterioration of the baby with increasing oxygen requirements and falling paO2, thoracotomy and bronchoplasty finally were performed on day 103 of life. The weight of the baby at time of surgery was 1,562 g.
RONCHIAL STENOSIS in children presents an even more challenging reconstructive procedure than in adults. The technical aspects of reconstruction in children may be similar to adults; however, they are more demanding in children given the smaller diameter of the airway. CASE REPORT A microprem baby (unique group of small infants who weigh less than 800 and have gestational ages ranging from 24 to 26 weeks have been called micro-preemies—Paediatric Surgery [ed 5] edited by James A. O’Neill, Jr. et al) delivered by emergency cesarian section at 26 weeks’ gestation was born with severe respiratory distress syndrome complicated with hyperbillirubinemia and bronchopulmonary dysplasia. The birth weight was 779 g. There was severe hyaline membrane disease that needed ventilation for 102 days. Patent ductus arteriosus was detected, which failed to respond to medical therapy. It was ligated on day 15 of life. After extubation, the child had collapse of the right lung. This did not respond to conservative measures of physiotherapy and postural drainage. Hence, a bronchoscopy was done that showed an annular stenosis of the right main bronchus with pin-hole opening. Trachea and left bronchus were normal. Tracheo-bronchography was not successful in delineating the airways (Fig 1). The stenosis could be dilated successfully with a fogarty catheter to about 40% size of lumen. However, 2 further attempts at dilatation did not show any improvement. A right pneumothorax occurred at the last attempt that failed to respond to the standard management of drainage of the pleural cavity.
From the Department of Paediatric Surgery, National University Hospital, Singapore. Address reprint requests to A/Prof K Prabhakaran, Chief, Department of Paediatric Surgery, Director, Liver Transplant Programme, National University Hospital, 5, Lower Kent Ridge Rd, Singapore 119074. © 2004 Elsevier Inc. All rights reserved. 1531-5037/04/3904-0037$30.00/0 doi:10.1016/j.jpedsurg.2003.12.031 e6
INDEX WORDS: Bronchial stenosis, bronchoplastic technique.
Surgical Technique Operative approach was from a right posterolateral thoracotomy through the fifth intercostal space. After initial exposure of the hilum and mobilization of the inferior pulmonary ligament, the right main bronchus with site of stenosis was exposed. An appropriate-size costal cartilage graft from the right sixth rib was harvested leaving the perichondrium intact. This was fashioned into a bridge and soaked in normal saline. Intraoperative bronchoscopy was used to determine the length of the stenosis. The endotracheal tube was inserted into the contralateral main-stem bronchus. The right main bronchus then was opened longitudinally along the stenotic segment (Fig 2). The costal cartilage patch was sutured to both the sides of the stenotic bronchus using fine running prolene sutures. The bronchopleural fistula track was then excised and repaired, and the tissue was sent for histology. Lung parenchyma was sutured with running sutures. The endotracheal tube was then pulled back above the carina, and another bronchoscopy was performed to access luminal patency. The chest was filled with saline to check for an air leak. The area was irrigated with antibiotic solution, and the repair was covered with parietal pleura to help seal the repair and increase vascularity. Two chest tubes were placed, and the chest was closed. Immediate improvement with good aeration of the collapsed lobe occurred. After the initial postoperative period, the general condition continued to improve with increased oral intake. The baby developed atelectasis on the right side on day 126 of life (postoperative day 23). However, this resolved spontaneously with good re-aeration of the whole of the right lung on day 130 of life. Need for oxygen decreased, and the baby was finally able to go off oxygen for the first time since birth, being comfortable on room air sucking well on the milk feedings. The baby was finally discharged on day 158 of life (postoperative day 55) with a weight of 3,770 g and length of 57 cm. The baby progressed well during the follow-up period. Growth was satisfactory. At last follow-up at the age of 10 years, weight was 26.5 kg and height was Journal of Pediatric Surgery, Vol 39, No 4 (April), 2004: E6-E8
BRONCHOPLASTY FOR NEONATAL BRONCHIAL STENOSIS
Fig 1. Tracheo-bronchography shows extravasation of dye into the thoracic cavity with failure to delineate the airways.
135.9cm and head circumference of 50.0 cm. Looking slightly slim, the child appears healthy and plays football and basketball. Chest x-ray done at the time was unremarkable (Fig 3).
DISCUSSION
Bronchial stenosis is an uncommon problem in children. Management can be difficult because of small luminal diameter and the proximity of the lung paren-
Fig 2. The hilum with stenotic segment of the right main bronchus exposed at surgery. The stenotic segment is longitudinally incised and a costal cartilage graft used for repair.
e7
Fig 3. Chest x-ray done at the age of 10 years is essentially unremarkable.
chyma to the bronchial lesion. Various methods of tracheobronchail reconstruction have been described with mixed results.1 Bronchoplastic procedures have obvious advantages in children because of their long life expectancy. The techniques allow reconstruction of the airway with maximal preservation of lung function.1 Long-term functional results are superior compared with pneumonectomy because of preserved parenchyma. Devastating complications such as postpneumonectomy syndrome are avoided.2 The technical aspects of reconstruction in children are similar to those used in adults; however, they are more demanding in children given the smaller size of airway. Clean transection lines, minimal handling of the mucosa, avoidance of devascularization, and precise placement of sutures are the basis of successful bronchoplastic techniques. Atelectasis has been reported as the most common complication after bronchoplastic procedures (2% to 20%). Technical errors in bronchial approximation, lymphatic interruption, and local postoperative edema may potentially contribute to atelectasis.3,4 Other common postoperative complications are stenosis (3% to 9%), postoperative pneumonia (4% to 6%), and empyema (2%).3,5,6 Careful mobilization without devascularization of the bronchus, taking care not to cause kinking, are crucial steps to avoid major complications.6 Bronchial stenosis in our case was most probably secondary to prolonged intubation, along with local inflammation, and unlikely to be congenital. Bronchial growth was not restricted, unlike that which is usually seen in congenital bronchial stenosis.7 In those cases, care should be taken in providing an intact bridge across the anterior bronchus, so that bronchial growth will not be restricted.7 A variety of stents are available to aid in the management of complex tracheal, carinal, and bronchial steno
e8
PRABHAKARAN AND TANKAR
sis. Complications are common, related to stent type and location, but can be managed with appropriate intervention.8 Our case highlights the successful application of bronchoplasty in a very small baby. Preoperative and intra-
operative bronchoscopy was helpful in localization of the endobronchial lesion. Understanding the principle, precise attention to technical detail and meticulous postoperative care, contribute toward long-term benefit for pulmonary conservation.
REFERENCES 1. Black CT, Luck SR, Raffensperger JG: Bronchoplastic techniques for pediatric lung salvage. J Pediatr Surg 23:653-656, 1988 2. Grillo HC, Shepard JAO, Mathisen DJ, et al: Postpneumonctomy syndrome: Diagnosis, management, and results. Ann Thorac Surg 54:638-651, 1992 3. Tedder M, Anstadt MP, Tedder SD, et al: Current morbidity, mortality, and survival after bronchoplastic procedures for malignancy. Ann Thorac Surg 54:387-391, 1992 4. Deslauriers J, Gaulin P, Beaulieu M, et al: Long-term clinical and functional results of sleeve lobectomy for primary lung cancer. J Thorac Cardiovasc Surg 92:871-879, 1986
5. Massard G, Kessler R, Gasser B, et al: Local control of disease and survival following bronchoplastic lobectomy for non-small cell lung cancer. Eur J Cardiothorac Surg 16:276-282, 1999 6. Muller C, Schinkel C, Hoffmann H, et al: Bronchoplstic resection for non-small cell lung cancer. Thorac Cardiovasc Surg 44:248-251, 1996 7. Bryant LR, Eiseman B: Replacement of tracheobronchial defects with autogenous pericardium. J Thoracic Cardiovasc Surg 48:733-740, 1964 8. Jacobs JP, Quintessenza JA, Botero LM, et al: The role of stents in the management of pediatric tracheal, carinal, and bronchial disease. Dur J Cardiothorac Surg 18:505-512, 2000
Bronchial stenosis is an uncommon problem in children. Management can be difficult because of the small luminal diameter and proximity of the lung parenchyma to the bronchial lesion. Bronchoplasty procedures have obvious advantages in children because of their long life expectancy. Longterm functional results are superior compared with pneumonectomy because of preserved lung tissue. A premature neonate weighing only 779 g at birth was born with severe respiratory distress syndrome complicated with hyperbillirubinemia and bronchopulmonary dysplasia. The child underwent bronchoplasty using a costal cartilage graft for a right main bronchus stenosis after 2 failed attempts at bronchoscopic dilatations. In spite of all complications, the
child improved sufficiently to be discharged without any oxygen dependency. Our case highlights the successful application of bronchoplasty in a very small baby. Preoperative and intraoperative bronchoscopy was helpful in localization of the endobronchial lesion. Understanding the principle, precise attention to technical detail and meticulous postoperative care contribute toward long-term benefit for pulmonary conservation. J Pediatr Surg 39:E6-E8. © 2004 Elsevier Inc. All rights reserved.
B
The baby had a bronchopleural fistula. In view of failure of all conservative measures and clinical deterioration of the baby with increasing oxygen requirements and falling paO2, thoracotomy and bronchoplasty finally were performed on day 103 of life. The weight of the baby at time of surgery was 1,562 g.
RONCHIAL STENOSIS in children presents an even more challenging reconstructive procedure than in adults. The technical aspects of reconstruction in children may be similar to adults; however, they are more demanding in children given the smaller diameter of the airway. CASE REPORT A microprem baby (unique group of small infants who weigh less than 800 and have gestational ages ranging from 24 to 26 weeks have been called micro-preemies—Paediatric Surgery [ed 5] edited by James A. O’Neill, Jr. et al) delivered by emergency cesarian section at 26 weeks’ gestation was born with severe respiratory distress syndrome complicated with hyperbillirubinemia and bronchopulmonary dysplasia. The birth weight was 779 g. There was severe hyaline membrane disease that needed ventilation for 102 days. Patent ductus arteriosus was detected, which failed to respond to medical therapy. It was ligated on day 15 of life. After extubation, the child had collapse of the right lung. This did not respond to conservative measures of physiotherapy and postural drainage. Hence, a bronchoscopy was done that showed an annular stenosis of the right main bronchus with pin-hole opening. Trachea and left bronchus were normal. Tracheo-bronchography was not successful in delineating the airways (Fig 1). The stenosis could be dilated successfully with a fogarty catheter to about 40% size of lumen. However, 2 further attempts at dilatation did not show any improvement. A right pneumothorax occurred at the last attempt that failed to respond to the standard management of drainage of the pleural cavity.
From the Department of Paediatric Surgery, National University Hospital, Singapore. Address reprint requests to A/Prof K Prabhakaran, Chief, Department of Paediatric Surgery, Director, Liver Transplant Programme, National University Hospital, 5, Lower Kent Ridge Rd, Singapore 119074. © 2004 Elsevier Inc. All rights reserved. 1531-5037/04/3904-0037$30.00/0 doi:10.1016/j.jpedsurg.2003.12.031 e6
INDEX WORDS: Bronchial stenosis, bronchoplastic technique.
Surgical Technique Operative approach was from a right posterolateral thoracotomy through the fifth intercostal space. After initial exposure of the hilum and mobilization of the inferior pulmonary ligament, the right main bronchus with site of stenosis was exposed. An appropriate-size costal cartilage graft from the right sixth rib was harvested leaving the perichondrium intact. This was fashioned into a bridge and soaked in normal saline. Intraoperative bronchoscopy was used to determine the length of the stenosis. The endotracheal tube was inserted into the contralateral main-stem bronchus. The right main bronchus then was opened longitudinally along the stenotic segment (Fig 2). The costal cartilage patch was sutured to both the sides of the stenotic bronchus using fine running prolene sutures. The bronchopleural fistula track was then excised and repaired, and the tissue was sent for histology. Lung parenchyma was sutured with running sutures. The endotracheal tube was then pulled back above the carina, and another bronchoscopy was performed to access luminal patency. The chest was filled with saline to check for an air leak. The area was irrigated with antibiotic solution, and the repair was covered with parietal pleura to help seal the repair and increase vascularity. Two chest tubes were placed, and the chest was closed. Immediate improvement with good aeration of the collapsed lobe occurred. After the initial postoperative period, the general condition continued to improve with increased oral intake. The baby developed atelectasis on the right side on day 126 of life (postoperative day 23). However, this resolved spontaneously with good re-aeration of the whole of the right lung on day 130 of life. Need for oxygen decreased, and the baby was finally able to go off oxygen for the first time since birth, being comfortable on room air sucking well on the milk feedings. The baby was finally discharged on day 158 of life (postoperative day 55) with a weight of 3,770 g and length of 57 cm. The baby progressed well during the follow-up period. Growth was satisfactory. At last follow-up at the age of 10 years, weight was 26.5 kg and height was Journal of Pediatric Surgery, Vol 39, No 4 (April), 2004: E6-E8
BRONCHOPLASTY FOR NEONATAL BRONCHIAL STENOSIS
Fig 1. Tracheo-bronchography shows extravasation of dye into the thoracic cavity with failure to delineate the airways.
135.9cm and head circumference of 50.0 cm. Looking slightly slim, the child appears healthy and plays football and basketball. Chest x-ray done at the time was unremarkable (Fig 3).
DISCUSSION
Bronchial stenosis is an uncommon problem in children. Management can be difficult because of small luminal diameter and the proximity of the lung paren-
Fig 2. The hilum with stenotic segment of the right main bronchus exposed at surgery. The stenotic segment is longitudinally incised and a costal cartilage graft used for repair.
e7
Fig 3. Chest x-ray done at the age of 10 years is essentially unremarkable.
chyma to the bronchial lesion. Various methods of tracheobronchail reconstruction have been described with mixed results.1 Bronchoplastic procedures have obvious advantages in children because of their long life expectancy. The techniques allow reconstruction of the airway with maximal preservation of lung function.1 Long-term functional results are superior compared with pneumonectomy because of preserved parenchyma. Devastating complications such as postpneumonectomy syndrome are avoided.2 The technical aspects of reconstruction in children are similar to those used in adults; however, they are more demanding in children given the smaller size of airway. Clean transection lines, minimal handling of the mucosa, avoidance of devascularization, and precise placement of sutures are the basis of successful bronchoplastic techniques. Atelectasis has been reported as the most common complication after bronchoplastic procedures (2% to 20%). Technical errors in bronchial approximation, lymphatic interruption, and local postoperative edema may potentially contribute to atelectasis.3,4 Other common postoperative complications are stenosis (3% to 9%), postoperative pneumonia (4% to 6%), and empyema (2%).3,5,6 Careful mobilization without devascularization of the bronchus, taking care not to cause kinking, are crucial steps to avoid major complications.6 Bronchial stenosis in our case was most probably secondary to prolonged intubation, along with local inflammation, and unlikely to be congenital. Bronchial growth was not restricted, unlike that which is usually seen in congenital bronchial stenosis.7 In those cases, care should be taken in providing an intact bridge across the anterior bronchus, so that bronchial growth will not be restricted.7 A variety of stents are available to aid in the management of complex tracheal, carinal, and bronchial steno
e8
PRABHAKARAN AND TANKAR
sis. Complications are common, related to stent type and location, but can be managed with appropriate intervention.8 Our case highlights the successful application of bronchoplasty in a very small baby. Preoperative and intra-
operative bronchoscopy was helpful in localization of the endobronchial lesion. Understanding the principle, precise attention to technical detail and meticulous postoperative care, contribute toward long-term benefit for pulmonary conservation.
REFERENCES 1. Black CT, Luck SR, Raffensperger JG: Bronchoplastic techniques for pediatric lung salvage. J Pediatr Surg 23:653-656, 1988 2. Grillo HC, Shepard JAO, Mathisen DJ, et al: Postpneumonctomy syndrome: Diagnosis, management, and results. Ann Thorac Surg 54:638-651, 1992 3. Tedder M, Anstadt MP, Tedder SD, et al: Current morbidity, mortality, and survival after bronchoplastic procedures for malignancy. Ann Thorac Surg 54:387-391, 1992 4. Deslauriers J, Gaulin P, Beaulieu M, et al: Long-term clinical and functional results of sleeve lobectomy for primary lung cancer. J Thorac Cardiovasc Surg 92:871-879, 1986
5. Massard G, Kessler R, Gasser B, et al: Local control of disease and survival following bronchoplastic lobectomy for non-small cell lung cancer. Eur J Cardiothorac Surg 16:276-282, 1999 6. Muller C, Schinkel C, Hoffmann H, et al: Bronchoplstic resection for non-small cell lung cancer. Thorac Cardiovasc Surg 44:248-251, 1996 7. Bryant LR, Eiseman B: Replacement of tracheobronchial defects with autogenous pericardium. J Thoracic Cardiovasc Surg 48:733-740, 1964 8. Jacobs JP, Quintessenza JA, Botero LM, et al: The role of stents in the management of pediatric tracheal, carinal, and bronchial disease. Dur J Cardiothorac Surg 18:505-512, 2000