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Lengthening technique for long gap esophageal atresia and early anastomosis
Lengthening Technique for Long Gap Esophageal Atresia and Early Anastomosis By Aayed R. Al-Qahtani, Salam Yazbeck, Nelson G. Rosen, Sami Youssef, and Sandeep K. Mayer Montreal, Quebec and Sherbrooke, Quebec
Background/Purpose: The treatment of long gap esophageal atresia remains a major surgical challenge. Whereas many approaches have been used for this problem, none are ideal. The authors used a technique originally described by Dr John E. Foker and accomplished early repair in 3 infants with long gap atresia. Methods: Three infants with esophageal atresia underwent thoracotomy shortly after birth and had a long gap preventing primary anastomosis. External traction sutures were placed on each esophageal pouch and exteriorized through the thoracic wall. The esophageal ends were approximated 1 to 2 mm daily by traction on the sutures. Anastomosis was performed when the 2 ends came together.
esophageal gaps were 3, 5, and 4.5 cm, respectively. Definitive anastomosis was performed at 14, 17, and 10 days, respectively. Two patients had anastomotic leaks that were treated conservatively. One patient had an esophageal stenosis that required dilatation. Conclusions: This technique allowed rapid esophageal lengthening in these 3 cases and led to early repair of long gap esophageal atresia, avoiding the need for a prolonged hospitalization or eventual replacement as well as long-term swallowing difficulties. J Pediatr Surg 38:737-739. © 2003 Elsevier Inc. All rights reserved.
Results: Three infants were included (31, 34, 37 weeks gestation, weights 1.38 kg, 1.9 kg, and 2.3 kg, respectively). The
INDEX WORDS: Esophageal atresia, long gap, lengthening technique.
L
Thoracotomy showed esophageal atresia with a proximal tracheoesophageal fistula. After fistula ligation and esophageal mobilization, primary anastomosis was impossible with an atretic gap of 4.5 cm. The traction technique was applied with 4-0 silk sutures and metallic clips placed on the sutures close to the esophageal ends to serve as radiopaque markers postoperatively. A gastrostomy also was performed. Daily traction of 1 to 2 mm on both esophageal ends was started on day 2 postoperatively. On day 7 a right pneumothorax with chylous effusion developed that resolved with drainage. On day 9, contrast studies of both esophageal pouches found that the ends were in proximity. A second thoracotomy was performed on day 10, and esophageal anastomosis was achieved with minimal tension. A contrast esophagram done one week post-anastomosis showed a small leak that responded well to conservative management. He was discharged on day 28 tolerating full oral feedings. At age
ONG GAP ESOPHAGEAL atresia remains an important challenge for pediatric surgeons. Many techniques have been described for the treatment of this problem, but none has proven ideal.1-8 Less than optimal results after esophageal replacement have led to a widely held belief that elongation of the native esophagus will provide a better functional outcome. Current elongation techniques may require a significant time delay until final repair, be associated with significant morbidity, and finally may fail to achieve the desired goal. Even when these techniques are successful, they often are followed by a long and difficult training period during which infants are unable to swallow. In 3 patients who presented with long gap esophageal atresias we used an esophageal lengthening technique originally described by Dr John E. Foker.9 This technique consists of the placement of traction sutures into the ends of the proximal and distal esophageal pouches after full mobilization and bringing these sutures together through the thoracic wall under slight tension (Figs 1 and 2). Postoperatively, these sutures are pulled 1 to 2 mm daily. Once the 2 ends of the esophagus are in proximity, esophageal anastomosis is performed. The details of each case are of sufficient importance to warrant individual presentation. CASE REPORTS
Case 1 A baby boy born at 37 weeks weighing 2,340 g had esophageal atresia without distal fistula diagnosed on day one of life. Journal of Pediatric Surgery, Vol 38, No 5 (May), 2003: pp 737-739
From the Hoˆpital Sainte-Justine, Universite´ de Montre´al, Montreal, Quebec, Canada, and the Universite´ de Sherbrooke, Sherbrooke, Quebec, Canada. Presented at the 34th Annual Meeting of the Canadian Association of Paediatric Surgeons, Vancouver, British Columbia, Canada, September 19-22, 2002. Address reprint requests to Dr Salam Yazbeck, Hoˆpital SainteJustine, 3175, Coˆte Sainte-Catherine, Montre´al, Que´bec, Canada H3T 1C5. © 2003 Elsevier Inc. All rights reserved. 0022-3468/03/3805-0020$30.00/0 10.1016/S0022-3468(03)00040-X 737
738
AL-QAHTANI ET AL
no leak but a small diverticulum at the anastomosis. Significant gastroesophageal reflux developed that eventually required fundoplication on day 63. Four months later he is doing well tolerating full oral feedings and gaining weight. Case 3 A boy born at 34 weeks weighing 1,955 g was transferred to our institution on day 4 of life with respiratory difficulty. Esophageal atresia with distal fistula was diagnosed, and thoracotomy was performed. After fistula ligation and esophageal mobilization, the atretic gap was 5 cm. Traction sutures of pledgeted 3-0 Dexon were placed with metallic clips on the suture ends. Postoperatively, traction of 1 mm daily was started on day 2. On day 9 a right chylothorax developed, which required tube drainage. On day 17 the proximal pouch sutures became loose and were withdrawn completely. A second thoracotomy was performed, and moderate inflammation was encountered with persistence of a significant gap between the ends. However, after extensive mobilization, an esophageal anastomosis was achieved under moderate tension. Postoperatively, an anastomotic leak without mediastinitis developed that was well controlled with
Fig 1. Schematic representation of the traction technique. The fixation of the distal pouch at the first thoracotomy is represented superiorly. Daily traction on the proximal pouch is shown inferiorly.
3 months he presented with a significant stenosis that required dilatation. He currently is one year old and doing well. Case 2 A 1,380-g boy was delivered vaginally at 31 weeks’ gestation after premature rupture of membranes. He was found to have esophageal atresia and distal tracheoesophageal fistula and on day one underwent thoracotomy. After fistula ligation and esophageal mobilization the atretic gap was 3 cm, and primary anastomosis was impossible. Traction sutures of 4-0 silk with teflon pledgets were placed with metallic clips placed on the sutures near each esophageal end. A gastrostomy also was performed. Traction of 1 to 2 mm daily began on day 2 and progressed until day 10 when the sutures in the distal pouch came out. Traction continued daily on the proximal pouch until the clips on the esophageal ends were in proximity on x-ray done on day 14. A small pneumothorax on day 4 resolved spontaneously. On day 14, a second thoracotomy was performed, and esophageal anastomosis was achieved with minimal tension. Contrast esophagogram on postoperative day 7 showed
Fig 2. Detail of traction suture placement in the esophageal pouches. Hemoclips are placed to identify the position of the pouches by radiograph during the traction process.
LENGTHENING TECHNIQUE FOR ESOPHAGEAL ATRESIA
739
drainage and resolved after 3 weeks. Follow-up after 4 months showed good weight gain, mild gastroesophageal reflux, and an asymptomatic moderate stable stricture at the anastomosis. He still has mild, but improving swallowing difficulty for which he requires intermittent gavages. DISCUSSION
Historically, the treatment of long gap esophageal atresia has involved initial cervical esophagostomy with eventual esophageal replacement.7,10,11 Suboptimal results with this strategy has led to techniques that try to elongate the esophagus enough to bridge the atretic gap. Such techniques have been described in various forms over the past 40 years.8,12-20 In a variety of tissues, including the esophagus, growth can be stimulated through tension and directional forces.19,21 This concept was the basis of the technique applied initially by Dr Foker on 4 patients with long gaps (5.5 to 6.8 cm) and in whom primary repair was achieved.9,22 Although this technique necessitates a second thoracotomy, the time required between the 2 is rather minimal when compared with other approaches. Long hospitalization and complications like aspiration also will be minimized by doing early anastomosis. In addition, swallowing difficulties for many months if not years to come probably will be avoided as well. As with any other new
technique, more adjustment is needed to achieve the best results with the least complications. Although the use of pledgeted sutures should tend to eliminate shear at the tissue-suture junction, both patients whose sutures were pledgeted had sutures cut through one of the esophageal pouches. Whereas the presence of the pledgets are not suspected to have caused the sutures to cut through, the optimal size, quantity, material, and placement technique for the traction sutures has yet to be determined. In case 3, moderate inflammation was found at the end of the esophageal pouch. Perhaps a less reactive suture or pledget material may minimize inflammation and further minimize complications. The original technique described placing the sutures extraluminally to avoid salivary leak and mediastinis.9 We suspect our sutures were placed transluminally. Whether this accounts for the pneumothoraces seen in 2 patients is uncertain. We believe also that placing the traction sutures transversely may decrease the risk of tearing through the esophageal pouches. Placing 2 clips on one side and one clip on the other side will make radiologic identification easier. The first stage of this 2-stage operation will possibly be amenable to a thoracoscopic approach in the future. Despite our limited experience with this technique, we feel it provides a superior alternative to many existing approaches for long gap atresia.
REFERENCES 1. Spitz L, Kiely EM, Drake DP, et al: Long-gap esophageal atresia. Pediatr Surg Int 11:462-465, 1996 2. Tsai J, Berkery L, Wesson D, et al: Esophageal atresia and tracheoesophageal fistula: A single institution’s experience over two decades. Ann Thorac Surg 64:778-783, 1997 3. Myers NA, Beasly SW, Auldist AW, et al: Esophageal atresia without fistula. Anastomosis or replacement. Pediatr Surg Int 2:216222, 1987 4. Spitz L: Esophageal atresia: Past, present, and future. J Pediatr Surg 31:19-25, 1996 5. Stringer MD: Esophageal substitution. Pediatr Surg Int 11:213, 1996 6. Rescorla FR, West KW, Scherer LR III, et al: The complex nature of type A (long-gap) esophageal atresia. Surgery 116:658-664, 1994 7. Ein SH, Shandling B, Stephens CA: Twenty-one year experience with the pediatric gastric tube. J Pediatr Surg 22:77-81, 1987 8. Howard R, Myers NA: Esophageal atresia: A technique for elongating the upper pouch. Surgery 58:725-727, 1965 9. Foker JE, Linden BC, Boyle EM Jr, et al: Development of a true primary repair for the full spectrum of esophageal atresia. Ann Surg 226:533-543, 1997 10. Hendren WH, Hendren WG: Colon interposition for esophagus in children. J Pediatr Surg 20:829-839, 1985 11. Saeki M, Tsuchida Y, Ogata T, et al: Long-term results of jejunal replacement of the esophagus. J Pediatr Surg 23:483-489, 1988 12. Thomasson BH: Congenital esophageal atresia: Mercury bag
stretching of the upper pouch in a patient without tracheoesophageal fistula. Surgery 71:661-664, 1972 13. Hendren WH, Hale JR: Esophageal atresia treated by electromagnetic bouginage and subsequent repair. J Pediatr Surg 11:713-722, 1976 14. Puri P, Khurana S: Delayed primary esophageal anastomosis for pure esophageal atresia. Semin Pediatr Surg 7:126-129, 1998 15. Livaditis A: Esophageal atresia: A method of overbridging large segmental gaps. Z Kinderchir 13:298-306, 1973 16. Eraklis AJ, Rosello PJ, Ballantine TVN: Circular esophagomyotomy of upper pouch in primary repair of long-segment esophageal atresia. J Pediatr Surg 11:709-712, 1976 17. Otte JB, Gianello P, Wese FX: Diverticulum formation after circular myotomy for esophageal atresia. J Pediatr Surg 19:68-71, 1984 18. Brown AK, Gough MH, Nicholls G, et al: Anterior flap repair of esophageal atresia: A 16-year evaluation. Pediatr Surg Int 10:525-528, 1995 19. Kimura K, Nishijima E, Tsugawa C, et al: Multistaged extrathoracic esophageal elongation procedure for long gap esophageal atresia: Experience with 12 patients. J Pediatr Surg 36:1725-1727, 2001 20. Lee H, Farmer D, Albanese C, et al: Traction elongation for treatment of long-gap esophageal atresia. Pediatr Endosurg Innovat Techniques 6:51-54, 2002 21. Ilizarov GA, Deviato AA: Surgical lengthening of the shin with simultaneous correction of deformities. Ortop Travmatol Protez 30:3237, 1969 22. Boyle EM Jr, Irwin ED, Foker JE: Primary repair of ultra-long gap esophageal atresia: Results without a lengthening procedure. Ann Thorac Surg 57:576-579, 1994
Background/Purpose: The treatment of long gap esophageal atresia remains a major surgical challenge. Whereas many approaches have been used for this problem, none are ideal. The authors used a technique originally described by Dr John E. Foker and accomplished early repair in 3 infants with long gap atresia. Methods: Three infants with esophageal atresia underwent thoracotomy shortly after birth and had a long gap preventing primary anastomosis. External traction sutures were placed on each esophageal pouch and exteriorized through the thoracic wall. The esophageal ends were approximated 1 to 2 mm daily by traction on the sutures. Anastomosis was performed when the 2 ends came together.
esophageal gaps were 3, 5, and 4.5 cm, respectively. Definitive anastomosis was performed at 14, 17, and 10 days, respectively. Two patients had anastomotic leaks that were treated conservatively. One patient had an esophageal stenosis that required dilatation. Conclusions: This technique allowed rapid esophageal lengthening in these 3 cases and led to early repair of long gap esophageal atresia, avoiding the need for a prolonged hospitalization or eventual replacement as well as long-term swallowing difficulties. J Pediatr Surg 38:737-739. © 2003 Elsevier Inc. All rights reserved.
Results: Three infants were included (31, 34, 37 weeks gestation, weights 1.38 kg, 1.9 kg, and 2.3 kg, respectively). The
INDEX WORDS: Esophageal atresia, long gap, lengthening technique.
L
Thoracotomy showed esophageal atresia with a proximal tracheoesophageal fistula. After fistula ligation and esophageal mobilization, primary anastomosis was impossible with an atretic gap of 4.5 cm. The traction technique was applied with 4-0 silk sutures and metallic clips placed on the sutures close to the esophageal ends to serve as radiopaque markers postoperatively. A gastrostomy also was performed. Daily traction of 1 to 2 mm on both esophageal ends was started on day 2 postoperatively. On day 7 a right pneumothorax with chylous effusion developed that resolved with drainage. On day 9, contrast studies of both esophageal pouches found that the ends were in proximity. A second thoracotomy was performed on day 10, and esophageal anastomosis was achieved with minimal tension. A contrast esophagram done one week post-anastomosis showed a small leak that responded well to conservative management. He was discharged on day 28 tolerating full oral feedings. At age
ONG GAP ESOPHAGEAL atresia remains an important challenge for pediatric surgeons. Many techniques have been described for the treatment of this problem, but none has proven ideal.1-8 Less than optimal results after esophageal replacement have led to a widely held belief that elongation of the native esophagus will provide a better functional outcome. Current elongation techniques may require a significant time delay until final repair, be associated with significant morbidity, and finally may fail to achieve the desired goal. Even when these techniques are successful, they often are followed by a long and difficult training period during which infants are unable to swallow. In 3 patients who presented with long gap esophageal atresias we used an esophageal lengthening technique originally described by Dr John E. Foker.9 This technique consists of the placement of traction sutures into the ends of the proximal and distal esophageal pouches after full mobilization and bringing these sutures together through the thoracic wall under slight tension (Figs 1 and 2). Postoperatively, these sutures are pulled 1 to 2 mm daily. Once the 2 ends of the esophagus are in proximity, esophageal anastomosis is performed. The details of each case are of sufficient importance to warrant individual presentation. CASE REPORTS
Case 1 A baby boy born at 37 weeks weighing 2,340 g had esophageal atresia without distal fistula diagnosed on day one of life. Journal of Pediatric Surgery, Vol 38, No 5 (May), 2003: pp 737-739
From the Hoˆpital Sainte-Justine, Universite´ de Montre´al, Montreal, Quebec, Canada, and the Universite´ de Sherbrooke, Sherbrooke, Quebec, Canada. Presented at the 34th Annual Meeting of the Canadian Association of Paediatric Surgeons, Vancouver, British Columbia, Canada, September 19-22, 2002. Address reprint requests to Dr Salam Yazbeck, Hoˆpital SainteJustine, 3175, Coˆte Sainte-Catherine, Montre´al, Que´bec, Canada H3T 1C5. © 2003 Elsevier Inc. All rights reserved. 0022-3468/03/3805-0020$30.00/0 10.1016/S0022-3468(03)00040-X 737
738
AL-QAHTANI ET AL
no leak but a small diverticulum at the anastomosis. Significant gastroesophageal reflux developed that eventually required fundoplication on day 63. Four months later he is doing well tolerating full oral feedings and gaining weight. Case 3 A boy born at 34 weeks weighing 1,955 g was transferred to our institution on day 4 of life with respiratory difficulty. Esophageal atresia with distal fistula was diagnosed, and thoracotomy was performed. After fistula ligation and esophageal mobilization, the atretic gap was 5 cm. Traction sutures of pledgeted 3-0 Dexon were placed with metallic clips on the suture ends. Postoperatively, traction of 1 mm daily was started on day 2. On day 9 a right chylothorax developed, which required tube drainage. On day 17 the proximal pouch sutures became loose and were withdrawn completely. A second thoracotomy was performed, and moderate inflammation was encountered with persistence of a significant gap between the ends. However, after extensive mobilization, an esophageal anastomosis was achieved under moderate tension. Postoperatively, an anastomotic leak without mediastinitis developed that was well controlled with
Fig 1. Schematic representation of the traction technique. The fixation of the distal pouch at the first thoracotomy is represented superiorly. Daily traction on the proximal pouch is shown inferiorly.
3 months he presented with a significant stenosis that required dilatation. He currently is one year old and doing well. Case 2 A 1,380-g boy was delivered vaginally at 31 weeks’ gestation after premature rupture of membranes. He was found to have esophageal atresia and distal tracheoesophageal fistula and on day one underwent thoracotomy. After fistula ligation and esophageal mobilization the atretic gap was 3 cm, and primary anastomosis was impossible. Traction sutures of 4-0 silk with teflon pledgets were placed with metallic clips placed on the sutures near each esophageal end. A gastrostomy also was performed. Traction of 1 to 2 mm daily began on day 2 and progressed until day 10 when the sutures in the distal pouch came out. Traction continued daily on the proximal pouch until the clips on the esophageal ends were in proximity on x-ray done on day 14. A small pneumothorax on day 4 resolved spontaneously. On day 14, a second thoracotomy was performed, and esophageal anastomosis was achieved with minimal tension. Contrast esophagogram on postoperative day 7 showed
Fig 2. Detail of traction suture placement in the esophageal pouches. Hemoclips are placed to identify the position of the pouches by radiograph during the traction process.
LENGTHENING TECHNIQUE FOR ESOPHAGEAL ATRESIA
739
drainage and resolved after 3 weeks. Follow-up after 4 months showed good weight gain, mild gastroesophageal reflux, and an asymptomatic moderate stable stricture at the anastomosis. He still has mild, but improving swallowing difficulty for which he requires intermittent gavages. DISCUSSION
Historically, the treatment of long gap esophageal atresia has involved initial cervical esophagostomy with eventual esophageal replacement.7,10,11 Suboptimal results with this strategy has led to techniques that try to elongate the esophagus enough to bridge the atretic gap. Such techniques have been described in various forms over the past 40 years.8,12-20 In a variety of tissues, including the esophagus, growth can be stimulated through tension and directional forces.19,21 This concept was the basis of the technique applied initially by Dr Foker on 4 patients with long gaps (5.5 to 6.8 cm) and in whom primary repair was achieved.9,22 Although this technique necessitates a second thoracotomy, the time required between the 2 is rather minimal when compared with other approaches. Long hospitalization and complications like aspiration also will be minimized by doing early anastomosis. In addition, swallowing difficulties for many months if not years to come probably will be avoided as well. As with any other new
technique, more adjustment is needed to achieve the best results with the least complications. Although the use of pledgeted sutures should tend to eliminate shear at the tissue-suture junction, both patients whose sutures were pledgeted had sutures cut through one of the esophageal pouches. Whereas the presence of the pledgets are not suspected to have caused the sutures to cut through, the optimal size, quantity, material, and placement technique for the traction sutures has yet to be determined. In case 3, moderate inflammation was found at the end of the esophageal pouch. Perhaps a less reactive suture or pledget material may minimize inflammation and further minimize complications. The original technique described placing the sutures extraluminally to avoid salivary leak and mediastinis.9 We suspect our sutures were placed transluminally. Whether this accounts for the pneumothoraces seen in 2 patients is uncertain. We believe also that placing the traction sutures transversely may decrease the risk of tearing through the esophageal pouches. Placing 2 clips on one side and one clip on the other side will make radiologic identification easier. The first stage of this 2-stage operation will possibly be amenable to a thoracoscopic approach in the future. Despite our limited experience with this technique, we feel it provides a superior alternative to many existing approaches for long gap atresia.
REFERENCES 1. Spitz L, Kiely EM, Drake DP, et al: Long-gap esophageal atresia. Pediatr Surg Int 11:462-465, 1996 2. Tsai J, Berkery L, Wesson D, et al: Esophageal atresia and tracheoesophageal fistula: A single institution’s experience over two decades. Ann Thorac Surg 64:778-783, 1997 3. Myers NA, Beasly SW, Auldist AW, et al: Esophageal atresia without fistula. Anastomosis or replacement. Pediatr Surg Int 2:216222, 1987 4. Spitz L: Esophageal atresia: Past, present, and future. J Pediatr Surg 31:19-25, 1996 5. Stringer MD: Esophageal substitution. Pediatr Surg Int 11:213, 1996 6. Rescorla FR, West KW, Scherer LR III, et al: The complex nature of type A (long-gap) esophageal atresia. Surgery 116:658-664, 1994 7. Ein SH, Shandling B, Stephens CA: Twenty-one year experience with the pediatric gastric tube. J Pediatr Surg 22:77-81, 1987 8. Howard R, Myers NA: Esophageal atresia: A technique for elongating the upper pouch. Surgery 58:725-727, 1965 9. Foker JE, Linden BC, Boyle EM Jr, et al: Development of a true primary repair for the full spectrum of esophageal atresia. Ann Surg 226:533-543, 1997 10. Hendren WH, Hendren WG: Colon interposition for esophagus in children. J Pediatr Surg 20:829-839, 1985 11. Saeki M, Tsuchida Y, Ogata T, et al: Long-term results of jejunal replacement of the esophagus. J Pediatr Surg 23:483-489, 1988 12. Thomasson BH: Congenital esophageal atresia: Mercury bag
stretching of the upper pouch in a patient without tracheoesophageal fistula. Surgery 71:661-664, 1972 13. Hendren WH, Hale JR: Esophageal atresia treated by electromagnetic bouginage and subsequent repair. J Pediatr Surg 11:713-722, 1976 14. Puri P, Khurana S: Delayed primary esophageal anastomosis for pure esophageal atresia. Semin Pediatr Surg 7:126-129, 1998 15. Livaditis A: Esophageal atresia: A method of overbridging large segmental gaps. Z Kinderchir 13:298-306, 1973 16. Eraklis AJ, Rosello PJ, Ballantine TVN: Circular esophagomyotomy of upper pouch in primary repair of long-segment esophageal atresia. J Pediatr Surg 11:709-712, 1976 17. Otte JB, Gianello P, Wese FX: Diverticulum formation after circular myotomy for esophageal atresia. J Pediatr Surg 19:68-71, 1984 18. Brown AK, Gough MH, Nicholls G, et al: Anterior flap repair of esophageal atresia: A 16-year evaluation. Pediatr Surg Int 10:525-528, 1995 19. Kimura K, Nishijima E, Tsugawa C, et al: Multistaged extrathoracic esophageal elongation procedure for long gap esophageal atresia: Experience with 12 patients. J Pediatr Surg 36:1725-1727, 2001 20. Lee H, Farmer D, Albanese C, et al: Traction elongation for treatment of long-gap esophageal atresia. Pediatr Endosurg Innovat Techniques 6:51-54, 2002 21. Ilizarov GA, Deviato AA: Surgical lengthening of the shin with simultaneous correction of deformities. Ortop Travmatol Protez 30:3237, 1969 22. Boyle EM Jr, Irwin ED, Foker JE: Primary repair of ultra-long gap esophageal atresia: Results without a lengthening procedure. Ann Thorac Surg 57:576-579, 1994