- Email: [email protected]
Closure of a benign bronchoesophageal fistula with endoscopic clips
Case Reports
In conclusion, endoscopic injection sclerotherapy with Histoacryl may be a good treatment modality in controlling large bleeding rectal varices. REFERENCES 1. Burroughs AK. The natural history of varices. J Hepatol 1993;17:S10-3. 2. Brewer TG. Treatment of acute gastroesophageal variceal hemorrhage. Med Clin North Am 1993;77:993-1014. 3. Pagliaro L, D’Amico G, Luca A, Pasta L, Politi F, Aragona E, et al. Portal hypertension: diagnosis and treatment. J Hepatol 1995;23:S36-44. 4. Chen WC, Hou MC, Lin HC, Chang FY, Lee SD. An endoscopic injection with N-butyl-2-cyanoacrylate used for colonic variceal bleeding: a case report and review of the literature. Am J Gastroenterol 2000; 95:540-2. 5. Hamlyn AN, Morris JS, Lunzer MR, Puritz H, Dick R. Portal hypertension with varices in unusual sites. Lancet 1974;28:1531-4. 6. Naveau S, Poynard T, Pauphilet C, Aubert A, Chaput JC. Rectal and colonic varices in cirrhosis [letter]. Lancet 1989;1:624. 7. Hosking SW, Smart HL, Johnson AG, Triger DR. Anorectal varices, haemorrhoids, and portal hypertension. Lancet 1989;1:349-52. 8. Kinkhabwala M, Mousavi A, Iyer S, Adamsons R. Bleeding ileal varicosity demonstrated by transhepatic portography. AJR Am J Roentgenol 1977;129:514-6. 9. Chawla Y, Dilawari JB. Anorectal varices: their frequency in cirrhotic and non-cirrhotic portal hypertension. Gut 1991;32:309-11. 10. Bresci G, Gambardella L, Parisi G, Federici G, Bertini M, Rindi G, et al. Colonic disease in cirrhotic patients with portal hypertension: an endoscopic and clinical evaluation. J Clin Gastroenterol 1998;26:222-7. 11. Khouqeer F, Morrow C, Jordan P. Duodenal varices as a cause of massive upper gastrointestinal bleeding. Surgery 1987;102:548-52. 12. Batoon SB, Zoneraich S. Misdiagnosed anorectal varices resulting in a fatal event. Am J Gastroenterol 1999;94:3076-7. 13. Herman BE, Baum S, Denobile J, Volpe RJ. Massive bleeding from rectal varices. Am J Gastroenterol 1993;88:939-42. 14. Waxman JS, Tarkin N, Dave P, Waxman M. Fatal hemorrhage from rectal varices. Report of two cases. Dis Colon Rectum 1984;27:749-50. 15. Firoozi B, Gamagaris Z, Weinshel EH, Bini EJ. Endoscopic band ligation of bleeding rectal varices. Dig Dis Sci 2002;47:1502-5. 16. Uno Y, Munakata A, Ishiguro A, Fukuda S, Sugai M, Munakata H. Endoscopic ligation for bleeding rectal varices in a child with primary extrahepatic portal hypertension. Endoscopy 1998;30:S107-8. 17. Levine J, Tahiri A, Banerjee B. Endoscopic ligation of bleeding rectal varices. Gastrointest Endosc 1993;39:188-90.
18. Yamanaka T, Shiraki K, Ito T, Sugimoto K, Sakai T, Ohmori S, et al. Endoscopic sclerotherapy (ethanolamine oleate injection) for acute rectal varices bleeding in a patient with liver cirrhosis. Hepatogastroenterology 2002;49:941-3. 19. Wang M, Desigan G, Dunn D. Endoscopic sclerotherapy for bleeding rectal varices: a case report. Am J Gastroenterol 1985;80:779-80. 20. Weiserbs DB, Zfass AM, Messmer J. Control of massive hemorrhage from rectal varices with sclerotherapy. Gastrointest Endosc 1986;32: 419-21. 21. Soehendra N, Grimm H, Nam VC, Berger B. N-butyl-2-cyanoacrylate: a supplement to endoscopic sclerotherapy. Endoscopy 1987;19:221-4. 22. Feretis C, Tabakopoulos D, Benakis P, Xenofontos M, Golematis B. Endoscopic hemostasis of esophageal and gastric variceal bleeding with Histoacryl. Endoscopy 1990;22:282-4. 23. Binmoeller KF, Soehendra N. Nonsurgical treatment of variceal bleeding: new modalities. Am J Gastroenterol 1995;90:1923-31. 24. D’Imperio N, Piemontese A, Baroncini D, Billi P, Borioni D, Dal Monte PP, et al. Evaluation of undiluted N-butyl-2-cyanoacrylate in the endoscopic treatment of upper gastrointestinal tract varices. Endoscopy 1996;28:239-43. 25. Huang YH, Yeh HZ, Chen GH, Chang CS, Wu CY, Poon SK, et al. Endoscopic treatment of bleeding gastric varices by N-butyl-2cyanoacrylate (Histoacryl) injection: long-term efficacy and safety. Gastrointest Endosc 2000;52:160-7. 26. Lo GH, Lai KH, Cheng JS, Chen MH, Chiang HT. A prospective, randomized trial of butyl cyanoacrylate injection versus band ligation in the management of bleeding gastric varices. Hepatology 2001;33: 1060-4. 27. Kim HG, Han KH, Lee CY, Chon CY, Moon YM, Kang JK, et al. Outcome of endoscopic injection therapy of Histoacryl in bleeding gastric varices [abstract]. Gastroenterology 1998;114:A1273. 28. Roesch W, Rexroth G. Pulmonary, cerebral and coronary emboli during bucrylate injection of bleeding fundic varices. Endoscopy 1998;30: S89-90. Current affiliations: Department of Internal Medicine, University of Inje College of Medicine, Seoul Paik Hospital, Seoul, Korea. Reprint requests: Jeong Seop Moon, MD, Department of Internal Medicine, University of Inje College of Medicine, Seoul Paik Hospital, 2-85 Jeo-dong, Joong-ku, 100-032, Seoul, Korea. Copyright ª 2005 by the American Society for Gastrointestinal Endoscopy 0016-5107/$30.00 doi:10.1016/j.gie.2005.05.012
Closure of a benign bronchoesophageal fistula with endoscopic clips Andrew Murdock, MRCP, R. J. Moorehead, FRCS, Tony C. K. Tham, FRCP Belfast, Northern Ireland, United Kingdom
Fistulas, although rare, may develop between the esophageal lumen and any other mediastinal structure. These tracheo- or bronchoesophageal fistulas can be divided into either congenital or acquired causes. Fistulas in adults are mostly acquired in nature, because it is rare for a congenital fistula to remain asymptomatic until adulthood. The majority of cases are caused by malignancy either
from a metastatic deposit or via direct invasion of a tumor. Benign causes also are seen and may be caused by a number of mechanisms. Infections, such as tuberculosis and histoplasmosis, can result in fistula formation, as can inflammatory conditions such as Crohn’s disease. Trauma from surgery or a prolonged period of intubation can predispose one to fistulas. The most common cause of
www.mosby.com/gie
Volume 62, No. 4 : 2005 GASTROINTESTINAL ENDOSCOPY 635
Case Reports
Figure 1. Fistula at base of ulcer.
benign tracheoesophageal fistulas is either a endotracheal or a tracheostomy tube. Diagnosis can present difficulties because of the nonspecific nature of the symptoms. Most patients are first seen with a pulmonary infection already present.1 We present a case of a benign bronchoesophageal fistula after esophagectomy for cancer treated with a combination of endoscopic clips and proton pump inhibitor (PPI) therapy. This technique can be used to avoid surgery.
CASE REPORT A 61-year-old man presented 8 years earlier with a 5month history of dysphagia for solids. His medical history of note included a perforated duodenal ulcer, which had been treated with a truncal vagotomy and gastroenterostomy 15 years earlier. A barium study and a EGD confirmed the presence of a tumor from 35 to 38 cm. Biopsies confirmed adenocarcinoma. A two-stage esophagectomy was performed. Histopathology showed an anaplastic carcinoma with clear resection margins. After a stormy recovery period in a high-dependency unit for adult respiratory distress syndrome, a 6-cycle course of chemotherapy was commenced. Good progress was noted over the next 4 years. Reflux, initially a problem, was controlled by standard doses of PPI therapy. During this period, he observed that if he drank without eating, he would cough for approximately 5 minutes afterward. Three years after his surgery, a barium swallow study was arranged. The study revealed passage of contrast into the right lower lobe bronchi occurring at the level of the anastomosis and suggested tumor recurrence. An EGD was performed, a fistula was found at 30 cm at the base of a deep ulcer (Fig. 1). No obvious evidence of tumor recurrence was seen. Biopsy specimens showed a mixture of inflammatory 636 GASTROINTESTINAL ENDOSCOPY Volume 62, No. 4 : 2005
Figure 2. Endoclip placement at fistula site.
cell infiltrate and granulation tissue. A CT of the chest and the abdomen was essentially normal. It was felt that as a result of his previous surgery, further operative intervention at the site of the fistula would be both difficult and hazardous because of problems with adhesions and the episode of life-threatening adult respiratory distress syndrome after his last operation. An EGD was performed with attempted placement of endoclips (Olympus America, Melville, NY) to close the fistula (Fig. 2). Clip placement was difficult, because the margins of the fistula were fibrotic. High-dose acid suppression was commenced with 30 mg lansoprazole twice a day. A review the following month showed some improvement in symptoms, but weight loss had become marked. A repeat study with water-soluble Gastromiro (Bracco, Milan, Italy) showed similar findings to before (Fig. 3), and a further EGD was arranged. At EGD, the previously seen fistula had reduced in size. Three further clips were applied to close the fistula. A repeat water-soluble Gastromiro study showed that the amount of contrast passing into the bronchial tree was significantly reduced from that seen previously. Over the next year, the patient remained well, gaining weight and not troubled by coughing during drinking or eating. Two years after treatment of his fistula, he reported a recurrence of reflux symptoms. A barium swallow did not show any fistula (Fig. 4), but a suspicious area at the anastomotic site was suggestive of recurrence. At EGD, a benign polyploid lesion was found at the site of the previous fistula (Fig. 5). Biopsies confirmed granulation tissue. No evidence of any fistula was visible.
DISCUSSION Benign bronchoesophageal fistulas can remain undiagnosed for years, because of a combination of their rarity and their nonspecific symptoms. Although there usually is www.mosby.com/gie
Case Reports
Figure 3. Gastromiro swallow, showing contrast entering bronchial tree.
a long interval before a diagnosis is made, there can be significant morbidity and mortality associated with the condition. Repeated infections can lead to bronchiectasis or abscess formation. A recent review of bronchoesophageal fistulas at Massachusetts General Hospital found that, in over 40 years, a total of 228 cases were seen.2 Benign causes made up only 6% of all cases. After esophageal surgery, 30% of benign causes developed. The majority of patients presented with a chronic cough; a mean length of time of symptoms was 7 years. Over 50% felt their cough worsened upon swallowing of fluid. In the vast majority of the cases, physical findings at examination were nonspecific in nature. Diagnostic options include barium swallow, bronchoscopy, EGD, and CT. CT with contrast can highlight the anatomy of the fistula tract. At endoscopy, the most common finding is of a papilla or a dimple. The most common fistula course found in the Massachusetts study was from the right bronchial tree to the distal esophagus (27-30 cm). This is similar to the course found in our case. For most cases of fistula formation, surgery is the traditional treatment. The fistula and any permanently damaged lung segments are removed. The standard operative procedure involves a thoracotomy, the fistula is exposed and divided, and both the defects in the bronchus and the esophagus are repaired with interposition of viable tissue between the suture lines. There are, however, a number of reports in the literature of endoscopic methods of attempting repair of fistulas. Local application of sodium hydroxide and acetic acid has been attempted.3 Application of fibrin glue also has been described to close fistulas in both adults and children.4-6 Fibrin glue is biocompatible, and so this treatment can be
repeated and does not inhibit any subsequent surgical intervention. Covered esophageal-wall-stent placement has been described for malignant fistulas7,8 associated with a stricture. ‘‘Double stenting,’’ i.e., insertion of both an esophageal and an airway stent, has been proposed as a superior option to stent placement of either the airway or the esophagus alone.9 Esophageal stent insertion would probably not have been suitable for our patient, because there was no stricture to hold the stent in place. A method proposed to prevent stent migration is the placement of
www.mosby.com/gie
Volume 62, No. 4 : 2005 GASTROINTESTINAL ENDOSCOPY 637
Figure 4. Barium study showing filling defect at site of anastomosis.
Figure 5. Benign polypoid lesion at site of previous fistula.
Case Reports
endoclips to act as an anchor for the stent.10,11 Presumably this may still not prevent stent migration if there is no luminal narrowing. The use of endoclips has been described for the treatment of both esophageal and gastric perforation. However, to our knowledge, there has been only one other description of the usage of endoclips in the successful closure of a benign esophageal fistula.12 In a case described by Mizobuchi et al,12 endoclips were used to close an esophagomediastinal-trachea fistula after failure of fibrin glue. Their report and now our case supports the use of endoclips to close benign esophageal fistulas as an alternative to surgery. REFERENCES 1. Gerzic Z, Rakic S, Randjelovic T. Acquired benign esophagorespiratory fistula: report of 16 consecutive cases. Ann Thorac Surg 1990;50:724-7. 2. Mangi A, Gaissert H, Wright C, Allan J, Wain J, Grillo H, et al. Benign broncho-esophageal fistula in the adult. Ann Thorac Surg 2002;73: 911-5. 3. Smith DC. A congenital broncho-esophageal fistula presenting in adult life without pulmonary infection. Br J Surg 1970;57:398-400. 4. Scappaticci E, Ardissone F, Baldi S, Coni F, Revello F, Filosso PL, et al. Closure of an iatrogenic tracheo-esophageal fistula with bronchoscopic gluing in a mechanically ventilated adult patient. Ann Thorac Surg 2004;77:328-9. 5. Kohler B, Kohler G, Riemann JF. Spontaneous esophagotracheal fistula resulting from ulcer in heterotrophic gastric mucosa. Gastroenterology 1988;95:828-30.
6. Ogunmola N, Wylie R, McDowell K, Kay M, Mahajan L. Endoscopic closure of esophagobronchial fistula with fibrin glue. J Pediatr Gastroenterol Nutr 2004;38:539-41. 7. Rajiman I. Endoscopic management of esophagorespiratory fistulas: expanding our options with expandable stents. Am J Gastroenterol 1998;93:496-9. 8. Dumonceau JM, Cremer M, Lalmand B, Deviere J. Esophageal fistula sealing: choice of stent, practical management, and cost. Gastrointest Endosc 1999;49:70-8. 9. Freitag L, Tekolf E, Steveling H, Donovan TJ, Stamatis G. Management of malignant esophagotracheal fistulas with airway stenting and double stenting. Chest 1996;110:1155-60. 10. Sriram PVJ, Das G, Rao GV, Reddy DN. Another novel use of endoscopic clipping: to anchor an esophageal endoprosthesis. Endoscopy 2001;33:724-6. 11. Silva RA, Dinis-Ribeiro M, Brandao C, Mesquita N, Fernandes N, LombaViana H, et al. Should we consider endoscopic clipping for prevention of stent migration? Endoscopy 2004;36:369-70. 12. Mizobuchi S, Kuge K, Maeda H, Matsumoto Y, Yamamoto M, Sasaguri S. Endoscopic clip application for closure of an esophagomediastinaltracheal fistula after surgery for esophageal cancer. Gastrointest Endosc 2003;57:962-5.
Current affiliations: Division of Gastroenterology and Gastrointestinal Surgery, Ulster Hospital, Dundonald, Belfast, Northern Ireland, UK. Reprint requests: A. Murdock, MRCP, Department of Gastroenterology, Ulster Hospital, Dundonald, Belfast BT16 1RH, Northern Ireland, UK. Copyright ª 2005 by the American Society for Gastrointestinal Endoscopy 0016-5107/$30.00 doi:10.1016/j.gie.2005.06.023
A case of toxic epidermal necrolysis with involvement of the GI tract after systemic contrast agent application at cardiac catheterization Austin Garza, MD, Adam J. Waldman, MD, Jay Mamel, MD, FACG, FACN Tampa, Florida, USA
CASE REPORT Our patient was a 62-year-old white man, who presented to The James A. Haley Veterans Hospital complaining of chest pain. He had a history of diabetes mellitus type 2, hyperlipidemia, peripheral vascular disease, chronic renal insufficiency, and significant alcohol abuse in the distant past. Allergies included penicillin but with an unknown reaction. A dipyridamole stress test revealed potential ischemia in the anterior, the inferior, and the inferolateral walls, with an ejection fraction of 55%. Subsequent outpatient cardiac catheterization revealed 3-vessel coronary artery disease, and his case was referred for surgical evaluation.
Two days after returning home from the procedure, his wife called his primary care physician stating that he had become ‘‘beet red, swollen all over, and hot one minute and cold the next, and shaking all over.’’ He was transported to the emergency department by emergency medical services. Eventually, some blistering of his skin developed. No treatment was administered, and this exanthem eventually resolved without further investigation. Based on his multivessel disease, he was deemed a good candidate for coronary artery bypass grafting. A presurgical, transesophageal echocardiogram revealed mild to moderate aortic stenosis, and valvuloplasty was tentatively scheduled for the same date. The patient agreed to proceed with the proposed plan to have surgical revascularization
638 GASTROINTESTINAL ENDOSCOPY Volume 62, No. 4 : 2005
www.mosby.com/gie
In conclusion, endoscopic injection sclerotherapy with Histoacryl may be a good treatment modality in controlling large bleeding rectal varices. REFERENCES 1. Burroughs AK. The natural history of varices. J Hepatol 1993;17:S10-3. 2. Brewer TG. Treatment of acute gastroesophageal variceal hemorrhage. Med Clin North Am 1993;77:993-1014. 3. Pagliaro L, D’Amico G, Luca A, Pasta L, Politi F, Aragona E, et al. Portal hypertension: diagnosis and treatment. J Hepatol 1995;23:S36-44. 4. Chen WC, Hou MC, Lin HC, Chang FY, Lee SD. An endoscopic injection with N-butyl-2-cyanoacrylate used for colonic variceal bleeding: a case report and review of the literature. Am J Gastroenterol 2000; 95:540-2. 5. Hamlyn AN, Morris JS, Lunzer MR, Puritz H, Dick R. Portal hypertension with varices in unusual sites. Lancet 1974;28:1531-4. 6. Naveau S, Poynard T, Pauphilet C, Aubert A, Chaput JC. Rectal and colonic varices in cirrhosis [letter]. Lancet 1989;1:624. 7. Hosking SW, Smart HL, Johnson AG, Triger DR. Anorectal varices, haemorrhoids, and portal hypertension. Lancet 1989;1:349-52. 8. Kinkhabwala M, Mousavi A, Iyer S, Adamsons R. Bleeding ileal varicosity demonstrated by transhepatic portography. AJR Am J Roentgenol 1977;129:514-6. 9. Chawla Y, Dilawari JB. Anorectal varices: their frequency in cirrhotic and non-cirrhotic portal hypertension. Gut 1991;32:309-11. 10. Bresci G, Gambardella L, Parisi G, Federici G, Bertini M, Rindi G, et al. Colonic disease in cirrhotic patients with portal hypertension: an endoscopic and clinical evaluation. J Clin Gastroenterol 1998;26:222-7. 11. Khouqeer F, Morrow C, Jordan P. Duodenal varices as a cause of massive upper gastrointestinal bleeding. Surgery 1987;102:548-52. 12. Batoon SB, Zoneraich S. Misdiagnosed anorectal varices resulting in a fatal event. Am J Gastroenterol 1999;94:3076-7. 13. Herman BE, Baum S, Denobile J, Volpe RJ. Massive bleeding from rectal varices. Am J Gastroenterol 1993;88:939-42. 14. Waxman JS, Tarkin N, Dave P, Waxman M. Fatal hemorrhage from rectal varices. Report of two cases. Dis Colon Rectum 1984;27:749-50. 15. Firoozi B, Gamagaris Z, Weinshel EH, Bini EJ. Endoscopic band ligation of bleeding rectal varices. Dig Dis Sci 2002;47:1502-5. 16. Uno Y, Munakata A, Ishiguro A, Fukuda S, Sugai M, Munakata H. Endoscopic ligation for bleeding rectal varices in a child with primary extrahepatic portal hypertension. Endoscopy 1998;30:S107-8. 17. Levine J, Tahiri A, Banerjee B. Endoscopic ligation of bleeding rectal varices. Gastrointest Endosc 1993;39:188-90.
18. Yamanaka T, Shiraki K, Ito T, Sugimoto K, Sakai T, Ohmori S, et al. Endoscopic sclerotherapy (ethanolamine oleate injection) for acute rectal varices bleeding in a patient with liver cirrhosis. Hepatogastroenterology 2002;49:941-3. 19. Wang M, Desigan G, Dunn D. Endoscopic sclerotherapy for bleeding rectal varices: a case report. Am J Gastroenterol 1985;80:779-80. 20. Weiserbs DB, Zfass AM, Messmer J. Control of massive hemorrhage from rectal varices with sclerotherapy. Gastrointest Endosc 1986;32: 419-21. 21. Soehendra N, Grimm H, Nam VC, Berger B. N-butyl-2-cyanoacrylate: a supplement to endoscopic sclerotherapy. Endoscopy 1987;19:221-4. 22. Feretis C, Tabakopoulos D, Benakis P, Xenofontos M, Golematis B. Endoscopic hemostasis of esophageal and gastric variceal bleeding with Histoacryl. Endoscopy 1990;22:282-4. 23. Binmoeller KF, Soehendra N. Nonsurgical treatment of variceal bleeding: new modalities. Am J Gastroenterol 1995;90:1923-31. 24. D’Imperio N, Piemontese A, Baroncini D, Billi P, Borioni D, Dal Monte PP, et al. Evaluation of undiluted N-butyl-2-cyanoacrylate in the endoscopic treatment of upper gastrointestinal tract varices. Endoscopy 1996;28:239-43. 25. Huang YH, Yeh HZ, Chen GH, Chang CS, Wu CY, Poon SK, et al. Endoscopic treatment of bleeding gastric varices by N-butyl-2cyanoacrylate (Histoacryl) injection: long-term efficacy and safety. Gastrointest Endosc 2000;52:160-7. 26. Lo GH, Lai KH, Cheng JS, Chen MH, Chiang HT. A prospective, randomized trial of butyl cyanoacrylate injection versus band ligation in the management of bleeding gastric varices. Hepatology 2001;33: 1060-4. 27. Kim HG, Han KH, Lee CY, Chon CY, Moon YM, Kang JK, et al. Outcome of endoscopic injection therapy of Histoacryl in bleeding gastric varices [abstract]. Gastroenterology 1998;114:A1273. 28. Roesch W, Rexroth G. Pulmonary, cerebral and coronary emboli during bucrylate injection of bleeding fundic varices. Endoscopy 1998;30: S89-90. Current affiliations: Department of Internal Medicine, University of Inje College of Medicine, Seoul Paik Hospital, Seoul, Korea. Reprint requests: Jeong Seop Moon, MD, Department of Internal Medicine, University of Inje College of Medicine, Seoul Paik Hospital, 2-85 Jeo-dong, Joong-ku, 100-032, Seoul, Korea. Copyright ª 2005 by the American Society for Gastrointestinal Endoscopy 0016-5107/$30.00 doi:10.1016/j.gie.2005.05.012
Closure of a benign bronchoesophageal fistula with endoscopic clips Andrew Murdock, MRCP, R. J. Moorehead, FRCS, Tony C. K. Tham, FRCP Belfast, Northern Ireland, United Kingdom
Fistulas, although rare, may develop between the esophageal lumen and any other mediastinal structure. These tracheo- or bronchoesophageal fistulas can be divided into either congenital or acquired causes. Fistulas in adults are mostly acquired in nature, because it is rare for a congenital fistula to remain asymptomatic until adulthood. The majority of cases are caused by malignancy either
from a metastatic deposit or via direct invasion of a tumor. Benign causes also are seen and may be caused by a number of mechanisms. Infections, such as tuberculosis and histoplasmosis, can result in fistula formation, as can inflammatory conditions such as Crohn’s disease. Trauma from surgery or a prolonged period of intubation can predispose one to fistulas. The most common cause of
www.mosby.com/gie
Volume 62, No. 4 : 2005 GASTROINTESTINAL ENDOSCOPY 635
Case Reports
Figure 1. Fistula at base of ulcer.
benign tracheoesophageal fistulas is either a endotracheal or a tracheostomy tube. Diagnosis can present difficulties because of the nonspecific nature of the symptoms. Most patients are first seen with a pulmonary infection already present.1 We present a case of a benign bronchoesophageal fistula after esophagectomy for cancer treated with a combination of endoscopic clips and proton pump inhibitor (PPI) therapy. This technique can be used to avoid surgery.
CASE REPORT A 61-year-old man presented 8 years earlier with a 5month history of dysphagia for solids. His medical history of note included a perforated duodenal ulcer, which had been treated with a truncal vagotomy and gastroenterostomy 15 years earlier. A barium study and a EGD confirmed the presence of a tumor from 35 to 38 cm. Biopsies confirmed adenocarcinoma. A two-stage esophagectomy was performed. Histopathology showed an anaplastic carcinoma with clear resection margins. After a stormy recovery period in a high-dependency unit for adult respiratory distress syndrome, a 6-cycle course of chemotherapy was commenced. Good progress was noted over the next 4 years. Reflux, initially a problem, was controlled by standard doses of PPI therapy. During this period, he observed that if he drank without eating, he would cough for approximately 5 minutes afterward. Three years after his surgery, a barium swallow study was arranged. The study revealed passage of contrast into the right lower lobe bronchi occurring at the level of the anastomosis and suggested tumor recurrence. An EGD was performed, a fistula was found at 30 cm at the base of a deep ulcer (Fig. 1). No obvious evidence of tumor recurrence was seen. Biopsy specimens showed a mixture of inflammatory 636 GASTROINTESTINAL ENDOSCOPY Volume 62, No. 4 : 2005
Figure 2. Endoclip placement at fistula site.
cell infiltrate and granulation tissue. A CT of the chest and the abdomen was essentially normal. It was felt that as a result of his previous surgery, further operative intervention at the site of the fistula would be both difficult and hazardous because of problems with adhesions and the episode of life-threatening adult respiratory distress syndrome after his last operation. An EGD was performed with attempted placement of endoclips (Olympus America, Melville, NY) to close the fistula (Fig. 2). Clip placement was difficult, because the margins of the fistula were fibrotic. High-dose acid suppression was commenced with 30 mg lansoprazole twice a day. A review the following month showed some improvement in symptoms, but weight loss had become marked. A repeat study with water-soluble Gastromiro (Bracco, Milan, Italy) showed similar findings to before (Fig. 3), and a further EGD was arranged. At EGD, the previously seen fistula had reduced in size. Three further clips were applied to close the fistula. A repeat water-soluble Gastromiro study showed that the amount of contrast passing into the bronchial tree was significantly reduced from that seen previously. Over the next year, the patient remained well, gaining weight and not troubled by coughing during drinking or eating. Two years after treatment of his fistula, he reported a recurrence of reflux symptoms. A barium swallow did not show any fistula (Fig. 4), but a suspicious area at the anastomotic site was suggestive of recurrence. At EGD, a benign polyploid lesion was found at the site of the previous fistula (Fig. 5). Biopsies confirmed granulation tissue. No evidence of any fistula was visible.
DISCUSSION Benign bronchoesophageal fistulas can remain undiagnosed for years, because of a combination of their rarity and their nonspecific symptoms. Although there usually is www.mosby.com/gie
Case Reports
Figure 3. Gastromiro swallow, showing contrast entering bronchial tree.
a long interval before a diagnosis is made, there can be significant morbidity and mortality associated with the condition. Repeated infections can lead to bronchiectasis or abscess formation. A recent review of bronchoesophageal fistulas at Massachusetts General Hospital found that, in over 40 years, a total of 228 cases were seen.2 Benign causes made up only 6% of all cases. After esophageal surgery, 30% of benign causes developed. The majority of patients presented with a chronic cough; a mean length of time of symptoms was 7 years. Over 50% felt their cough worsened upon swallowing of fluid. In the vast majority of the cases, physical findings at examination were nonspecific in nature. Diagnostic options include barium swallow, bronchoscopy, EGD, and CT. CT with contrast can highlight the anatomy of the fistula tract. At endoscopy, the most common finding is of a papilla or a dimple. The most common fistula course found in the Massachusetts study was from the right bronchial tree to the distal esophagus (27-30 cm). This is similar to the course found in our case. For most cases of fistula formation, surgery is the traditional treatment. The fistula and any permanently damaged lung segments are removed. The standard operative procedure involves a thoracotomy, the fistula is exposed and divided, and both the defects in the bronchus and the esophagus are repaired with interposition of viable tissue between the suture lines. There are, however, a number of reports in the literature of endoscopic methods of attempting repair of fistulas. Local application of sodium hydroxide and acetic acid has been attempted.3 Application of fibrin glue also has been described to close fistulas in both adults and children.4-6 Fibrin glue is biocompatible, and so this treatment can be
repeated and does not inhibit any subsequent surgical intervention. Covered esophageal-wall-stent placement has been described for malignant fistulas7,8 associated with a stricture. ‘‘Double stenting,’’ i.e., insertion of both an esophageal and an airway stent, has been proposed as a superior option to stent placement of either the airway or the esophagus alone.9 Esophageal stent insertion would probably not have been suitable for our patient, because there was no stricture to hold the stent in place. A method proposed to prevent stent migration is the placement of
www.mosby.com/gie
Volume 62, No. 4 : 2005 GASTROINTESTINAL ENDOSCOPY 637
Figure 4. Barium study showing filling defect at site of anastomosis.
Figure 5. Benign polypoid lesion at site of previous fistula.
Case Reports
endoclips to act as an anchor for the stent.10,11 Presumably this may still not prevent stent migration if there is no luminal narrowing. The use of endoclips has been described for the treatment of both esophageal and gastric perforation. However, to our knowledge, there has been only one other description of the usage of endoclips in the successful closure of a benign esophageal fistula.12 In a case described by Mizobuchi et al,12 endoclips were used to close an esophagomediastinal-trachea fistula after failure of fibrin glue. Their report and now our case supports the use of endoclips to close benign esophageal fistulas as an alternative to surgery. REFERENCES 1. Gerzic Z, Rakic S, Randjelovic T. Acquired benign esophagorespiratory fistula: report of 16 consecutive cases. Ann Thorac Surg 1990;50:724-7. 2. Mangi A, Gaissert H, Wright C, Allan J, Wain J, Grillo H, et al. Benign broncho-esophageal fistula in the adult. Ann Thorac Surg 2002;73: 911-5. 3. Smith DC. A congenital broncho-esophageal fistula presenting in adult life without pulmonary infection. Br J Surg 1970;57:398-400. 4. Scappaticci E, Ardissone F, Baldi S, Coni F, Revello F, Filosso PL, et al. Closure of an iatrogenic tracheo-esophageal fistula with bronchoscopic gluing in a mechanically ventilated adult patient. Ann Thorac Surg 2004;77:328-9. 5. Kohler B, Kohler G, Riemann JF. Spontaneous esophagotracheal fistula resulting from ulcer in heterotrophic gastric mucosa. Gastroenterology 1988;95:828-30.
6. Ogunmola N, Wylie R, McDowell K, Kay M, Mahajan L. Endoscopic closure of esophagobronchial fistula with fibrin glue. J Pediatr Gastroenterol Nutr 2004;38:539-41. 7. Rajiman I. Endoscopic management of esophagorespiratory fistulas: expanding our options with expandable stents. Am J Gastroenterol 1998;93:496-9. 8. Dumonceau JM, Cremer M, Lalmand B, Deviere J. Esophageal fistula sealing: choice of stent, practical management, and cost. Gastrointest Endosc 1999;49:70-8. 9. Freitag L, Tekolf E, Steveling H, Donovan TJ, Stamatis G. Management of malignant esophagotracheal fistulas with airway stenting and double stenting. Chest 1996;110:1155-60. 10. Sriram PVJ, Das G, Rao GV, Reddy DN. Another novel use of endoscopic clipping: to anchor an esophageal endoprosthesis. Endoscopy 2001;33:724-6. 11. Silva RA, Dinis-Ribeiro M, Brandao C, Mesquita N, Fernandes N, LombaViana H, et al. Should we consider endoscopic clipping for prevention of stent migration? Endoscopy 2004;36:369-70. 12. Mizobuchi S, Kuge K, Maeda H, Matsumoto Y, Yamamoto M, Sasaguri S. Endoscopic clip application for closure of an esophagomediastinaltracheal fistula after surgery for esophageal cancer. Gastrointest Endosc 2003;57:962-5.
Current affiliations: Division of Gastroenterology and Gastrointestinal Surgery, Ulster Hospital, Dundonald, Belfast, Northern Ireland, UK. Reprint requests: A. Murdock, MRCP, Department of Gastroenterology, Ulster Hospital, Dundonald, Belfast BT16 1RH, Northern Ireland, UK. Copyright ª 2005 by the American Society for Gastrointestinal Endoscopy 0016-5107/$30.00 doi:10.1016/j.gie.2005.06.023
A case of toxic epidermal necrolysis with involvement of the GI tract after systemic contrast agent application at cardiac catheterization Austin Garza, MD, Adam J. Waldman, MD, Jay Mamel, MD, FACG, FACN Tampa, Florida, USA
CASE REPORT Our patient was a 62-year-old white man, who presented to The James A. Haley Veterans Hospital complaining of chest pain. He had a history of diabetes mellitus type 2, hyperlipidemia, peripheral vascular disease, chronic renal insufficiency, and significant alcohol abuse in the distant past. Allergies included penicillin but with an unknown reaction. A dipyridamole stress test revealed potential ischemia in the anterior, the inferior, and the inferolateral walls, with an ejection fraction of 55%. Subsequent outpatient cardiac catheterization revealed 3-vessel coronary artery disease, and his case was referred for surgical evaluation.
Two days after returning home from the procedure, his wife called his primary care physician stating that he had become ‘‘beet red, swollen all over, and hot one minute and cold the next, and shaking all over.’’ He was transported to the emergency department by emergency medical services. Eventually, some blistering of his skin developed. No treatment was administered, and this exanthem eventually resolved without further investigation. Based on his multivessel disease, he was deemed a good candidate for coronary artery bypass grafting. A presurgical, transesophageal echocardiogram revealed mild to moderate aortic stenosis, and valvuloplasty was tentatively scheduled for the same date. The patient agreed to proceed with the proposed plan to have surgical revascularization
638 GASTROINTESTINAL ENDOSCOPY Volume 62, No. 4 : 2005
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