- Email: [email protected]
Delayed fistulisation from esophageal replacement surgery
Seminars in Pediatric Surgery (2009) 18, 104-108
Delayed fistulisation from esophageal replacement surgery Ahmed Abdalwahab, Mohammed Al Namshan, Abdullah Al Rabeeah, Jean-Martin Laberge, MD, FRCSC, FACS From the Section of Pediatric Surgery, King AbdulAziz Medical City, Jeddah, Kingdom of Saudi Arabia. KEYWORDS Esophageal replacement; Gastric tube; Pericardial fistula; Gastric pull-up; Gastro-bronchial fistula; Complications
We present two patients who developed delayed fistulisation following esophageal replacement surgery. The first is a 13-year-old child who, at the age of 3 years, underwent a trans-mediastinal colonic esophageal replacement for a refractory corrosive injury followed by a retrosternal reverse gastric tube after an early catastrophic leak. Ten years later, he presented with a history of intermittent chest pain for 6 months. He developed a tension pneumopericardial tamponade caused by a fistula between gastric tube and pericardium. He recovered after sternotomy. The second was born prematurely with type C esophageal atresia and other malformations. After esophageal anastomosis, he developed a refractory stricture that was resected at 10 months. Despite a fundoplication at 4 years, the recurrent esophageal stricture required resection at 14 years, accomplished by mobilizing the stomach into the chest through a left thoracoabdominal incision. The postoperative course was complicated by a gastric leak in the chest with empyema, but the patient recovered and was able to eat. Five years later, he underwent an anterior spinal fusion to correct a worsening kyphoscoliosis. Postoperatively, he developed an ARDS picture, leakage of air through the gastrostomy, and a fatal pulmonary hemorrhage secondary to a gastrobronchial fistula. Fistulisation from esophageal replacement surgery represents a rare long-term complication that pediatric surgeons need to be aware of. © 2009 Elsevier Inc. All rights reserved.
Esophageal replacement for refractory corrosive injuries and esophageal atresia using colonic interposition, gastric tubes, and gastric pull-up has been used by pediatric surgeons for decades.1-7 These have been placed transmediastinal, retrosternal, subcutaneously, and even transpleurally,8 depending on the clinical situation and the surgeons’ practice. We present unusual long-term complications in two children, one with a retrosternal reverse gastric tube in whom a pericardial fistula occurred more than 10 years after the procedure, and the other with a gastrobronchial fistula that developed 5 years after a gastric pull-up. Address reprint requests and correspondence: Ahmed Abdalwahab, King AbdulAziz Medical City–Jeddah, PO Box 9515, Jeddah 21423, Kingdom of Saudi Arabia. E-mail address: [email protected].
1055-8586/$ -see front matter © 2009 Elsevier Inc. All rights reserved. doi:10.1053/j.sempedsurg.2009.02.008
Case 1 NG is a 14-year-old male who, at the age of 3 years, suffered an esophageal corrosive injury. He was treated initially conservatively but developed a complex stricture refractory to dilation. He underwent a transmediastinal colonic esophageal replacement with early failure due to a massive anastamotic leak and was managed with a cervical esophagostomy. He later underwent a successful retrosternal reversed stapled gastric tube replacement by the senior author (AR). He was followed for the next 4 years and then was lost to follow-up. He was reportedly doing well for the 7 intervening years until the current symptoms appeared. He presented to the internal medicine service in a community hospital with a 6-month history of intermittent left chest
Abdalwahab, Al Namshan, and Al Rabeeah
Esophageal Replacement Surgery
Figure 1 PA radiograph of the chest shows a left pleural effusion and barium in the pericardial sac.
pain, generalized aches, and occasional vomiting. The pain increased for the last 10 days with mild shortness of breath. A chest x-ray (CXR) showed a large cardiac shadow. An echocardiogram showed a small to moderate pericardial effusion. He was started on intravenous antibiotics and steroids with a provisional diagnosis of autoimmune pericarditis and was admitted to the intensive care unit, with a plan to perform pericardiocentesis as a part of the medical work-up. Due to his surgical history, there was concern not to cause injury to the gastric tube during pericardiocentesis; therefore, a barium swallow was performed. Contrast was seen in the pericardium. He was then transferred to KAMC/Jed for further evaluation and treatment. On presentation, he was afebrile, he-
105
modynamically stable, and continued to have mild chest pain with shortness of breath. The steroids were stopped, intravenous Esomeprazole (Nexium; AstraZeneca, Södertälje, Sweden) was started. and the antibiotics modified. A chest radiograph (Figure 1) showed a left pleural effusion, a large cardiac silhouette, and barium lining the pericardium. A chest tube was inserted into the left pleural cavity and drained 250 mL of serous fluid. The echocardiogram was repeated showing minimal pericardial effusion with possible pericardial calcifications, which probably was the barium. A CT scan of the chest showed air and barium in the pericardium with close proximity of the gastric tube to the superior mediastinal vessels and heart. A water-soluble contrast esophageal swallow was done to define the anatomy for surgical intervention, and this clearly defined the fistula between the gastric tube and pericardium (Figure 2). Soon after, he became febrile, tachycardic, and tachypneic. The chest tube drained 400 mL of purulent fluid, and the patient became more dyspnoeic requiring intubation. This resulted in hemodynamic instability managed by vasopressors. A repeat CXR showed pneumopericardium (Figure 3). An urgent CT chest was performed with aspiration of about 30 mL of air from the pericardium, but we failed to introduce an indwelling catheter. The patient promptly improved, came off pressor support, and was extubated. He was transferred to KAMC/RIY via Medevac with cabin pressure precautions for cardiac surgery support and further management. After further stabilization, a sternotomy was performed. The gastric tube was preserved, and the fistula was identified and ligated. A biopsy was taken from the edge. The pericardium was found to have food remnants and was washed out (Figure 4). A pericardial flap interposition was performed as the fistula occurred at the staple line. There was no evidence of ulceration on inspection of the tube, and passage of a Hegar dilator showed no stricture distally. A
Figure 2 Contrast esophagram. (A) Lateral view of the chest demonstrates the retrosternal gastric tube with the fistula connecting to the pericardial sac. (B) The AP view shows the pericardium full of contrast.
106
Figure 3 A lateral view of the chest shows a pneumopericardium mixed with old contrast.
gastrostomy tube was placed with gastrojejunal tube advancement for postoperative feeding. The postoperative course was uneventful, and the patient stayed in the ICU for 5 days. The postoperative contrast swallow showed no leak. Slow oral feedings were started, and the chest tube was removed. He was discharged 2 weeks postoperatively. The pathology showed marked inflammation with no evidence of ulceration. At 20 months postoperation, he remains well on oral Omperazole (Apo-omeprazole; Apotex Inc., Weston, Canada).
Seminars in Pediatric Surgery, Vol 18, No 2, May 2009 stricture on contrast swallow. At 14 years of age, a left thoracoabdominal approach was used to resect the esophageal stricture, mobilize the distal esophagus and stomach up through the hiatus to allow a primary esophagoesophagostomy; a Belsey-type fundoplication was done as well as a gastrostomy and a feeding jejunostomy. The resected esophageal stricture demonstrated areas of respiratory epithelium, cartilage and bronchial glands on microscopic analysis, as well as gastric-type mucosa and signs of reflux esophagitis. The postoperative course was complicated, with a gastric perforation in the chest requiring two reoperations for repair and pulmonary decortication. Toward the end of this admission, he underwent two esophageal dilatations and a final one 8 months later. At the age of 19 years, the patient underwent anterior spinal fusion through a right thoracoabdominal approach for the correction of a kyphoscoliosis. One of the main complaints was worsening of a right retrosternal pain over the previous 2 years, which seemed related to worsening of the kyphosis. Postoperatively, the patient developed left lower lobe atelectasis and hypercapnia; he required reintubation because of a shock/ARDS picture and bleeding from an erosive esophagitis. Over the next few days, while remaining in critical condition, a lot of air was noted coming out from the gastrostomy tube. This was thought to be caused by overflow from the high ventilator pressures required to treat the ARDS; the possibility of reopening of the tracheoesophageal fistula closed 19 years earlier was raised but thought unlikely. Ten days after surgery, a copious amount of fresh blood came from the endotracheal tube. A flexible bronchoscopy showed fresh blood coming from the left lower lobe bronchus, and chest radiograph demonstrated intraparenchymal barium from an esophagram done the day before. The bleeding was controlled with a Blakemore tube, but the patient’s pulmonary condition continued to deterio-
Case 2 This patient was born prematurely, weighting 1.6 kg with EATEF, duodenal atresia, and hemivertebrae. At birth, he underwent duodeno-duodenostomy and gastric division with a double gastrostomy— one to drain the upper third of the stomach, and the other to enable feeding in the distal two-thirds. The EATEF was repaired at 2 months of age, and the stomach was reanastomosed 2 months later. He developed a severe esophageal stricture that became impossible to dilate and was treated with resection and reanastomosis at 10 months of life. The patient required multiple esophageal dilatations over the ensuing years, yet the gastrostomy was closed at 2 years. At 4 years of age, after an episode of esophageal foreign body impaction, a Nissen fundoplication was done as well as a gastrostomy and placement of a string to allow retrograde dilatations. Despite multiple dilatations, the patient could only take clear fluids by mouth and appeared to have a 7-cm-long
Figure 4 An operative view with the patient’s head toward the left; the retrosternal gastric tube is encircled with a penrose drain proximally and distally and is rotated to show the nasogastric tube protruding from the fistula opening; the pericardium is in the lower portion of the picture. (Color version of figure is available online.)
Abdalwahab, Al Namshan, and Al Rabeeah
Esophageal Replacement Surgery
rate and he died. Autopsy demonstrated gastric ulcers, erosive gastroesophagitis, and a gastrobronchial fistula involving a segmental bronchus of the left lower lobe, with eroded and thrombosed arteries from the stomach and left lower lobe.
Discussion Esophageal pericardial fistulas are a rare occurrence. They have been reported in the adult population as spontaneous or conduit fistulas with underlying malignancy,9,10 radiation,11 gastric ulceration,12-14 tuberculosis,15 and after laparoscopic fundoplication.16 In the pediatric population, esophageal conduit fistulas have been mostly reported as complications of ulcers secondary to gastric reflux into the conduit and have been reported up to 21 years after the operation.17 Symptoms of these fistulas range widely according to the organs involved; Debras and coworkers successfully treated a 22-year-old man who developed an aortocolonic fistula secondary to an ulcer 21 years after interposition for esophageal atresia.17 A patient with a left ventriculocolic fistula occurring 12 years after colonic replacement for caustic injury presented with recurrent hematemesis that proved difficult to diagnose and treat after multiple interventions.18 Colopericardial,19,20 colobronchial,11 and gastric tube to left interlobar pulmonary artery fistulas21 have also been reported. The variety of major vascular and mediastinal structures involved illustrate the challenge in managing these complications and highlights the need to work closely with cardiac surgeons and have cardiac bypass back-up available. Salo and coworkers describe a child similar to our first case who developed a retrosternal gastric tube to pericardial fistula (after a failed transmediastinal colonic interposition) secondary to Barrett’s esophagus. The patient presented with severe mediastinitis and was managed with diversion and later subcutaneous ileocolic interposition.22 The symptoms also vary widely according to the organ involved. Cough, dysphagia, hemoptysis, dyspnoea, recurrent respiratory infections, and hematemesis are commonly reported symptoms. The diagnosis can be highly aided by contrast studies or CT scans. We have not found echocardiogram helpful in our first case. Acute pneumopericardium occurred in our first patient after intubation, and he developed tamponade with positive pressure ventilation. This has been described without tamponade in two adult cases with a large gastroesophageal ulcer eroding into the pericardium and successfully managed surgically.14,23 Several authors have used upper GI endoscopy intraoperatively for diagnosis.14,21,24 Due to our first patient’s presentation, we elected against performing endoscopy. In the second patient, endoscopy had been performed in the ICU and only showed erosive esophagitis. The pathology of the fistula edges in our reversed gastric tube case showed no ulceration. Despite that, many reports
107
indicated ulceration secondary to reflux to be a major cause in fistulisation and advocate keeping these children on longterm prophylaxis. In the second case, ulcerations were prominent and occurred despite aggressive medical antireflux treatment. Attempts to treat these fistulas medically have failed.18,21,25 In conclusion, fistulisation into the pericardium or segmental bronchus after esophageal replacement surgery is a very rare complication that pediatric surgeons need to be aware of. Long-term close follow-up is required for early diagnosis and prompt surgical management.
References 1. Borgnon J, Tounian P, Auber F, et al. Esophageal replacement in children by an isoperistaltic gastric tube: a 12-year experience. Pediatr Surg Int 2004;20:829-33. 2. Appignani A, Lauro V, Pretipino M, et al. Intestinal bypass of the oesophagus: 117 patients in 28 years. Pediatric Surg Int 2000;16: 326-8. 3. Ein SH. Gastric tubes in children with caustic esophageal injury: a 32-year review. J Pediatr Surg 1998;33:1363-5. 4. Khan AR, Stiff G, Mohammed AR, et al. Esophageal replacement with colon in children. Pediatr Surg Int 1998;13:79-83. 5. Spitz L, Kiely E, Pierro A. Gastric transposition in children: a 21-year experience. J Pediatr Surg 2004;39:276-81. 6. Tannuri U, Maksoud-Filho JG, Tannuri AC, et al. Which is better for esophageal substitution in children, esophagocoloplasty or gastric transposition? A 27-year experience of a single center. J Pediatr Surg 2007;42:500-4. 7. Hamza AF, Abdelhay S, Sherif H, et al. Caustic esophageal strictures in children: 30 years’ experience. J Pediatr Surg 2003;38: 828-33. 8. Gounot E, Borgnon J, Huet F, et al. Isolated isoperistaltic gastric tube interposition for esophageal replacement in children. J Pediatr Surg 2006;41:592-5. 9. Schouten van der Velden AP, Ruers TJ, Bonenkamp JJA. Cardiogastric fistula after gastric tube interposition. A case report and review of literature. J Surg Oncol 2007;95:79-82. 10. Kaufman J, Thongsuwan N, Stern E, et al. Esophageal-pericardial fistula with purulent pericarditis secondary to esophageal carcinoma presenting with tamponade. Ann Thorac Surg 2003;75:288-9. 11. Zhao X, Sandhu B, Kiev J. Colobronchial fistula as a rare complication of coloesophageal interposition: a unique treatment with a review of the medical literature. Am Surg 2005;71:1058-9. 12. Chapman PR, Boals JR. Pneumopericardium caused by giant gastric ulcer. AJR Am J Roentgenol 1998;171:1669-70. 13. Porteous C, Williams D, Foulis A, et al. Penetration of the left ventricular myocardium by benign peptic ulceration: two cases and a review of the published work. J Clin Pathol 1984;37:1239-44. 14. Cozart JC, Sundaresan S, Chokshi HR, et al. Gastropericardial fistula with pneumopericardium: an unusual complication of benign peptic ulceration. Gastrointest Endosc 1999;49:387-90. 15. Al-Ajmi J, Al-Soub H, El-Deeb Y. Pyopneumopericardium due to esophago-pericardial fistula in patient with tuberculous pericarditis. Saudi Med J 2007;28:969-70. 16. Sihvo EI, Räsänen JV, Hynninen M, et al. Gastropericardial fistula, purulent pericarditis, and cardiac tamponade after laparoscopic Nissen fundoplication. Ann Thorac Surg 2006;81:356-8. 17. Debras B, Kanane O, Enon B, et al. Aorto-colonic fistula as a late complication of colon interposition for oesophageal atresia. Eur J Pediatr Surg 1996;6:310-11.
108 18. Pantelides ML, Fitzgerald MD. Left ventriculo-colic fistula–a late complication of colonic interposition for the oesophagus. Postgrad Med J 1988;64:710-12. 19. Wetstein L, Ergin MA, Griepp RB. Colo-pericardial fistula: complication of colonic interposition. Tex Heart Inst J 1982;9: 373-6. 20. Huddleston DJ, Kocoshis TA. Death in custody due to a colopericardial fistula. Am J Forensic Med Pathol 1997;18:194-8. 21. Hazebroek EJ, Hazebroek FW, Leibman S, et al. Late presentation of gastric tube ulcer perforation after oesophageal atresia repair. Pediatr Surg Int 2008;24:869-71.
Seminars in Pediatric Surgery, Vol 18, No 2, May 2009 22. Salo JA, Heikkilä L, Nemlander A, et al. Barrett’s oesophagus and perforation of gastric tube ulceration into the pericardium: a late complication after reconstruction of oesophageal atresia. Ann Chir Gynaecol 1995;84:92-4. 23. Müller AM, Betz MJ, Kromeier J, et al. Images in cardiovascular medicine. Acute pneumopericardium due to intestino-pericardial fistula. Circulation 2006;114:e7-9. 24. Schneider F, Schenk M, Tempé JD, et al. Spontaneous gastropericardial fistula. Ann Emerg Med 1995;26:394. 25. Osaki T, Matsuura H. Thoracic empyema and lung abscess resulting from gastropulmonary fistula as a complication of esophagectomy. Ann Thorac Cardiovasc Surg 2008;14:172-4.
Delayed fistulisation from esophageal replacement surgery Ahmed Abdalwahab, Mohammed Al Namshan, Abdullah Al Rabeeah, Jean-Martin Laberge, MD, FRCSC, FACS From the Section of Pediatric Surgery, King AbdulAziz Medical City, Jeddah, Kingdom of Saudi Arabia. KEYWORDS Esophageal replacement; Gastric tube; Pericardial fistula; Gastric pull-up; Gastro-bronchial fistula; Complications
We present two patients who developed delayed fistulisation following esophageal replacement surgery. The first is a 13-year-old child who, at the age of 3 years, underwent a trans-mediastinal colonic esophageal replacement for a refractory corrosive injury followed by a retrosternal reverse gastric tube after an early catastrophic leak. Ten years later, he presented with a history of intermittent chest pain for 6 months. He developed a tension pneumopericardial tamponade caused by a fistula between gastric tube and pericardium. He recovered after sternotomy. The second was born prematurely with type C esophageal atresia and other malformations. After esophageal anastomosis, he developed a refractory stricture that was resected at 10 months. Despite a fundoplication at 4 years, the recurrent esophageal stricture required resection at 14 years, accomplished by mobilizing the stomach into the chest through a left thoracoabdominal incision. The postoperative course was complicated by a gastric leak in the chest with empyema, but the patient recovered and was able to eat. Five years later, he underwent an anterior spinal fusion to correct a worsening kyphoscoliosis. Postoperatively, he developed an ARDS picture, leakage of air through the gastrostomy, and a fatal pulmonary hemorrhage secondary to a gastrobronchial fistula. Fistulisation from esophageal replacement surgery represents a rare long-term complication that pediatric surgeons need to be aware of. © 2009 Elsevier Inc. All rights reserved.
Esophageal replacement for refractory corrosive injuries and esophageal atresia using colonic interposition, gastric tubes, and gastric pull-up has been used by pediatric surgeons for decades.1-7 These have been placed transmediastinal, retrosternal, subcutaneously, and even transpleurally,8 depending on the clinical situation and the surgeons’ practice. We present unusual long-term complications in two children, one with a retrosternal reverse gastric tube in whom a pericardial fistula occurred more than 10 years after the procedure, and the other with a gastrobronchial fistula that developed 5 years after a gastric pull-up. Address reprint requests and correspondence: Ahmed Abdalwahab, King AbdulAziz Medical City–Jeddah, PO Box 9515, Jeddah 21423, Kingdom of Saudi Arabia. E-mail address: [email protected].
1055-8586/$ -see front matter © 2009 Elsevier Inc. All rights reserved. doi:10.1053/j.sempedsurg.2009.02.008
Case 1 NG is a 14-year-old male who, at the age of 3 years, suffered an esophageal corrosive injury. He was treated initially conservatively but developed a complex stricture refractory to dilation. He underwent a transmediastinal colonic esophageal replacement with early failure due to a massive anastamotic leak and was managed with a cervical esophagostomy. He later underwent a successful retrosternal reversed stapled gastric tube replacement by the senior author (AR). He was followed for the next 4 years and then was lost to follow-up. He was reportedly doing well for the 7 intervening years until the current symptoms appeared. He presented to the internal medicine service in a community hospital with a 6-month history of intermittent left chest
Abdalwahab, Al Namshan, and Al Rabeeah
Esophageal Replacement Surgery
Figure 1 PA radiograph of the chest shows a left pleural effusion and barium in the pericardial sac.
pain, generalized aches, and occasional vomiting. The pain increased for the last 10 days with mild shortness of breath. A chest x-ray (CXR) showed a large cardiac shadow. An echocardiogram showed a small to moderate pericardial effusion. He was started on intravenous antibiotics and steroids with a provisional diagnosis of autoimmune pericarditis and was admitted to the intensive care unit, with a plan to perform pericardiocentesis as a part of the medical work-up. Due to his surgical history, there was concern not to cause injury to the gastric tube during pericardiocentesis; therefore, a barium swallow was performed. Contrast was seen in the pericardium. He was then transferred to KAMC/Jed for further evaluation and treatment. On presentation, he was afebrile, he-
105
modynamically stable, and continued to have mild chest pain with shortness of breath. The steroids were stopped, intravenous Esomeprazole (Nexium; AstraZeneca, Södertälje, Sweden) was started. and the antibiotics modified. A chest radiograph (Figure 1) showed a left pleural effusion, a large cardiac silhouette, and barium lining the pericardium. A chest tube was inserted into the left pleural cavity and drained 250 mL of serous fluid. The echocardiogram was repeated showing minimal pericardial effusion with possible pericardial calcifications, which probably was the barium. A CT scan of the chest showed air and barium in the pericardium with close proximity of the gastric tube to the superior mediastinal vessels and heart. A water-soluble contrast esophageal swallow was done to define the anatomy for surgical intervention, and this clearly defined the fistula between the gastric tube and pericardium (Figure 2). Soon after, he became febrile, tachycardic, and tachypneic. The chest tube drained 400 mL of purulent fluid, and the patient became more dyspnoeic requiring intubation. This resulted in hemodynamic instability managed by vasopressors. A repeat CXR showed pneumopericardium (Figure 3). An urgent CT chest was performed with aspiration of about 30 mL of air from the pericardium, but we failed to introduce an indwelling catheter. The patient promptly improved, came off pressor support, and was extubated. He was transferred to KAMC/RIY via Medevac with cabin pressure precautions for cardiac surgery support and further management. After further stabilization, a sternotomy was performed. The gastric tube was preserved, and the fistula was identified and ligated. A biopsy was taken from the edge. The pericardium was found to have food remnants and was washed out (Figure 4). A pericardial flap interposition was performed as the fistula occurred at the staple line. There was no evidence of ulceration on inspection of the tube, and passage of a Hegar dilator showed no stricture distally. A
Figure 2 Contrast esophagram. (A) Lateral view of the chest demonstrates the retrosternal gastric tube with the fistula connecting to the pericardial sac. (B) The AP view shows the pericardium full of contrast.
106
Figure 3 A lateral view of the chest shows a pneumopericardium mixed with old contrast.
gastrostomy tube was placed with gastrojejunal tube advancement for postoperative feeding. The postoperative course was uneventful, and the patient stayed in the ICU for 5 days. The postoperative contrast swallow showed no leak. Slow oral feedings were started, and the chest tube was removed. He was discharged 2 weeks postoperatively. The pathology showed marked inflammation with no evidence of ulceration. At 20 months postoperation, he remains well on oral Omperazole (Apo-omeprazole; Apotex Inc., Weston, Canada).
Seminars in Pediatric Surgery, Vol 18, No 2, May 2009 stricture on contrast swallow. At 14 years of age, a left thoracoabdominal approach was used to resect the esophageal stricture, mobilize the distal esophagus and stomach up through the hiatus to allow a primary esophagoesophagostomy; a Belsey-type fundoplication was done as well as a gastrostomy and a feeding jejunostomy. The resected esophageal stricture demonstrated areas of respiratory epithelium, cartilage and bronchial glands on microscopic analysis, as well as gastric-type mucosa and signs of reflux esophagitis. The postoperative course was complicated, with a gastric perforation in the chest requiring two reoperations for repair and pulmonary decortication. Toward the end of this admission, he underwent two esophageal dilatations and a final one 8 months later. At the age of 19 years, the patient underwent anterior spinal fusion through a right thoracoabdominal approach for the correction of a kyphoscoliosis. One of the main complaints was worsening of a right retrosternal pain over the previous 2 years, which seemed related to worsening of the kyphosis. Postoperatively, the patient developed left lower lobe atelectasis and hypercapnia; he required reintubation because of a shock/ARDS picture and bleeding from an erosive esophagitis. Over the next few days, while remaining in critical condition, a lot of air was noted coming out from the gastrostomy tube. This was thought to be caused by overflow from the high ventilator pressures required to treat the ARDS; the possibility of reopening of the tracheoesophageal fistula closed 19 years earlier was raised but thought unlikely. Ten days after surgery, a copious amount of fresh blood came from the endotracheal tube. A flexible bronchoscopy showed fresh blood coming from the left lower lobe bronchus, and chest radiograph demonstrated intraparenchymal barium from an esophagram done the day before. The bleeding was controlled with a Blakemore tube, but the patient’s pulmonary condition continued to deterio-
Case 2 This patient was born prematurely, weighting 1.6 kg with EATEF, duodenal atresia, and hemivertebrae. At birth, he underwent duodeno-duodenostomy and gastric division with a double gastrostomy— one to drain the upper third of the stomach, and the other to enable feeding in the distal two-thirds. The EATEF was repaired at 2 months of age, and the stomach was reanastomosed 2 months later. He developed a severe esophageal stricture that became impossible to dilate and was treated with resection and reanastomosis at 10 months of life. The patient required multiple esophageal dilatations over the ensuing years, yet the gastrostomy was closed at 2 years. At 4 years of age, after an episode of esophageal foreign body impaction, a Nissen fundoplication was done as well as a gastrostomy and placement of a string to allow retrograde dilatations. Despite multiple dilatations, the patient could only take clear fluids by mouth and appeared to have a 7-cm-long
Figure 4 An operative view with the patient’s head toward the left; the retrosternal gastric tube is encircled with a penrose drain proximally and distally and is rotated to show the nasogastric tube protruding from the fistula opening; the pericardium is in the lower portion of the picture. (Color version of figure is available online.)
Abdalwahab, Al Namshan, and Al Rabeeah
Esophageal Replacement Surgery
rate and he died. Autopsy demonstrated gastric ulcers, erosive gastroesophagitis, and a gastrobronchial fistula involving a segmental bronchus of the left lower lobe, with eroded and thrombosed arteries from the stomach and left lower lobe.
Discussion Esophageal pericardial fistulas are a rare occurrence. They have been reported in the adult population as spontaneous or conduit fistulas with underlying malignancy,9,10 radiation,11 gastric ulceration,12-14 tuberculosis,15 and after laparoscopic fundoplication.16 In the pediatric population, esophageal conduit fistulas have been mostly reported as complications of ulcers secondary to gastric reflux into the conduit and have been reported up to 21 years after the operation.17 Symptoms of these fistulas range widely according to the organs involved; Debras and coworkers successfully treated a 22-year-old man who developed an aortocolonic fistula secondary to an ulcer 21 years after interposition for esophageal atresia.17 A patient with a left ventriculocolic fistula occurring 12 years after colonic replacement for caustic injury presented with recurrent hematemesis that proved difficult to diagnose and treat after multiple interventions.18 Colopericardial,19,20 colobronchial,11 and gastric tube to left interlobar pulmonary artery fistulas21 have also been reported. The variety of major vascular and mediastinal structures involved illustrate the challenge in managing these complications and highlights the need to work closely with cardiac surgeons and have cardiac bypass back-up available. Salo and coworkers describe a child similar to our first case who developed a retrosternal gastric tube to pericardial fistula (after a failed transmediastinal colonic interposition) secondary to Barrett’s esophagus. The patient presented with severe mediastinitis and was managed with diversion and later subcutaneous ileocolic interposition.22 The symptoms also vary widely according to the organ involved. Cough, dysphagia, hemoptysis, dyspnoea, recurrent respiratory infections, and hematemesis are commonly reported symptoms. The diagnosis can be highly aided by contrast studies or CT scans. We have not found echocardiogram helpful in our first case. Acute pneumopericardium occurred in our first patient after intubation, and he developed tamponade with positive pressure ventilation. This has been described without tamponade in two adult cases with a large gastroesophageal ulcer eroding into the pericardium and successfully managed surgically.14,23 Several authors have used upper GI endoscopy intraoperatively for diagnosis.14,21,24 Due to our first patient’s presentation, we elected against performing endoscopy. In the second patient, endoscopy had been performed in the ICU and only showed erosive esophagitis. The pathology of the fistula edges in our reversed gastric tube case showed no ulceration. Despite that, many reports
107
indicated ulceration secondary to reflux to be a major cause in fistulisation and advocate keeping these children on longterm prophylaxis. In the second case, ulcerations were prominent and occurred despite aggressive medical antireflux treatment. Attempts to treat these fistulas medically have failed.18,21,25 In conclusion, fistulisation into the pericardium or segmental bronchus after esophageal replacement surgery is a very rare complication that pediatric surgeons need to be aware of. Long-term close follow-up is required for early diagnosis and prompt surgical management.
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