Cas cliniques Fistule aorto-oesophagienne secondaire a la mise en place d’un stent oesophagien : Traitement en urgence par cyanoacrylate et stentgraft Moonsang Ahn,1 Byung Seok Shin,2 Mi-hyun Park,3 Taejon et Chungnam, Coree du Sud
Nous rapportons l’embolisation par N-butyl 2-cyanoacrylate suivie de la mise en place endovasculaire d’un stentgraft pour le traitement d’une fistule aorto-œsophagienne secondaire au tait admis placement d’un stent oesophagien. Un homme de 53 ans avec un cancer du poumon e mate me se massive due a la formation d’une fistule aorto-œsophagienne 20 jours avec une he s le placement d’un stent oesophagien. L’injection du N-butyl 2-cyanoacrylate dans la fistule apre te effectue e en tant que traitement d’urgence devant un e tat aorto-oesophagienne a e modynamique instable, et un stentgraft endovasculaire a e te place ulte rieurement par he diaire de l’arte re fe morale droite. Le patient allait bien sans he mate me se jusqu’a ce qu’il l’interme meure de pneumonie 45 jours plus tard. L’embolisation par cyanoacrylate et le placement d’un morragie massive provoque e par une fistule aorto-oesophagienne est stentgraft pour traiter l’he thode rapide et efficace et peut e ^tre une alternative a la re paration chirurgicale. une me
Aortoesophageal fistula (AEF) is a rare, usually fatal condition that causes massive hematemesis. AEF caused by an esophageal stent is extremely rare; only a few cases have been reported.1-5 To the best of our knowledge, this is the first report of the successful treatment of AEF secondary to esophageal stent placement with N-butyl 2-cyanoacrylate (NBCA) embolization and subsequent endovascular stent graft placement.
DOI of original article: 10.1016/j.avsg.2009.12.009. 1 Department of Surgery, Chungnam National University Hospital, Taejon, Cor ee du Sud. 2 Department of Radiology, Chungnam National University Hospital, Taejon, Cor ee du Sud. 3 Department of Radiology, Dankook University Hospital, Chungnam, Coree du Sud.
Correspondence : Byung Seok Shin, Department of Radiology, Chungnam National University Hospital, 640 Daesa-Dong, Taejon, 301-721, Coree du Sud, E-mail:
[email protected] Ann Vasc Surg 2010; 24: 555.e1-555.e5 DOI: 10.1016/j.acvfr.2010.12.042 Ó Annals of Vascular Surgery Inc. e par ELSEVIER MASSON SAS Edit
CASE REPORT A 53-year-old man with lung cancer was admitted with dysphagia. He had undergone radiation and chemotherapy 8 months earlier. Endoscopy revealed extrinsic esophageal compression, and metastatic lymphadenopathy compressing the esophagus and left atrium was observed on computed tomography (CT) scanning. An 18-mmdiameter, 10-cm-long, retrievable covered esophageal stent (CHOOSTENT; M.I. Tech, Seoul, Korea) (Fig. 1A) was placed in the mid-esophagus by an endoscopist. The patient was admitted with massive hematemesis 20 days after esophageal stent placement. Endoscopy identified massive bleeding at the proximal stent margin, suggesting the presence of an AEF. We performed emergency angiography, which showed contrast leakage into the esophageal lumen around the esophageal stent due to the fistula (Fig. 2A). The patient became hypotensive and entered a state of hypovolemic shock despite ongoing fluid resuscitation. During resuscitation, which included intubation and cardiac massage, we decided on embolization with NBCA (Histoacryl ; B. Braun, Melsungen, Germany) for prompt emergent treatment. After direct catheterization with a 5-F Cobra catheter (Cook, Bloomington, IN) into the fistula orifice, a 3-F microcatheter (Cook) was advanced through the AEF. After the
604.e17
604.e18 Cas cliniques
Annales de chirurgie vasculaire
Fig. 1. Covered esophageal stent and self-expandable thoracic stent graft. A Covered esophageal stent (CHOOSTENT; M.I. Tech, Seoul, Korea) consists of a nitinol wire and polyurethane membrane. B Thoracic stent graft (SEAL Thoracic Flex Stent Graft; S&G Biotech INC, Seongnam-Si, Korea) is a domestic device approved for use in Korea that consists of a nitinol wire and polyester (Dacron). microcatheter was flushed with 5% dextrose in water, we injected 1 mL of NBCA mixed with ethiodol (Lipiodol; Savage Laboratories, Melville, NY) (1:1) into the fistula under fluoroscopy; the microcatheter was then flushed again with 5% dextrose in water. Using fluoroscopy, we observed that the injected NBCA migrated to the more distal portion of the fistula. We believe that migration of NBCA was caused by high aortic pressure and, therefore, additional injections would be required for adequate occlusion of the AEF. While injecting NBCA from esophageal side of the fistula, we draw the microcatheter very slowly and cautiously into the fistula to prevent unwanted regurgitation into the aortic lumen. Finally, we filled the NBCA into the fistula without regurgitation into the aorta. A total of 4 mL NBCA was required to completely obliterate the fistula (Fig. 2B). The injections took a total of 10 minutes. Immediately after embolization, the patient’s hemodynamics became stable, and no further leakage of contrast was observed on angiography. We decided to cover the AEF by placing an endovascular stent graft. Under general anesthesia, a 32-mm 10cm self-expandable thoracic stent graft (SEAL Thoracic Flex Stent Graft ; S&G Biotech INC, Seongnam-Si, Korea) (Fig. 1B) was advanced into the thoracic aorta via the right femoral artery. After placement of the endovascular stent graft, no perigraft leakage or filling of the fistula tract was observed on the aortogram (Fig. 3). No complications
Fig. 2. A Angiogram shows contrast extravasation into the esophageal lumen through an aortoesophageal fistula tract at the level of the proximal end of the esophageal stent. B After embolization with NBCA, a radiopaque material is filled around the esophageal stent. occurred during or after the procedure. Antibiotic therapy for possible bacteremia and transient mild fever was initiated on the day of stent grafting. These symptoms resolved after 3 days, but antibiotics were continued throughout the patient’s course of treatment. Follow-up CT scan 2 days after stent grafting showed high attenuation between the proximal portion of the esophageal stent and the aortic stent graft, indicative of NBCA in the AEF (Fig. 4). No further hematemesis occurred. The patient was discharged in a stable condition 12 days after the stent graft, and no further bleeding associated with AEF
Vol. 24, No. 4, 2010
Cas cliniques 604.e19
Fig. 3. After endovascular stent graft placement was performed, no perigraft contrast leakage was observed on aortography. occurred. The patient died of pneumonia 45 days after stent grafting.
DISCUSSION An AEF is a life-threatening condition due to massive bleeding, and it requires prompt recognition and management. An AEF is usually caused by descending thoracic aortic diseases, including aneurysm, foreign body ingestion, and esophageal diseases such as esophageal cancer or ulcers. Among them, thoracic aortic aneurysm is responsible for approximately two-thirds of AEFs. Rapid diagnosis and repair to reduce morbidity and mortality are important in the management of AEFs. Previous reports recommend prompt open surgical repair or emergent endovascular stent graft.6-15 An AEF caused by an esophageal stent is extremely rare. Only a few cases have been reported, and there was only one case of successful surgical repair of AEF with patient survival (Table I). According to previous reports, hematemesis usually occurs within 5 weeks of esophageal stent placement, and most patients died within 36 hours after admission for hematemesis. Most reported cases of AEFs associated with esophageal stents, including our patient, typically occurred around the proximal portion of the esophageal stent. Four reports described perforation or erosion of the proximal end of the stent into the aorta at autopsy or emergent thoracotomy.1-5 To the best of our knowledge, this is the first report of successful NBCA
Fig. 4. CT scans obtained 2 days after stent placement show high-attenuation embolic material (arrow) between the esophageal stent and aortic stent graft on axial A and sagittal B scans. No signs suggesting infection, such as air bubbles or fluid collection around the fistula, were observed.
embolization and subsequent endovascular stent graft placement for treatment of an AEF secondary to placement of an esophageal stent. We attempted NBCA embolization for AEF because the patient developed hypovolemic shock during angiography due to massive bleeding, which had to be controlled immediately in the angiography suite. Bleeding was controlled by NBCA embolization within 10 minutes.
Doing well, 3 monthsafter operation Died Fail
Wound infection PyothoraxGraft infection (e) . Success
Um et al.5 (2009)
Unosawa et al.4 (2008)
Esophageal cancer Esophageal strictureafter irradiation forlymphoma Benign esophagealstricture and ulcercaused by refluxesophagitis Esophageal strictureafter chemoirradiationfor lung cancer Siersema et al.2 (1997) Rogary et al.3 (2007)
Graft replacementof the descending aortaEsophagectomy Angiography
Died Died . . Fail Fail
Died . Esophageal cancer Allgaier el al.1 (1997)
Endoscopic intervention Sengstaken-Blakemore tube EndoscopyTransfusion Emergency thoracotomy
Fail
Follow-up/mortality Cause for esophageal stent
Management of AEF
Result
Complication
Annales de chirurgie vasculaire
Authors/year
Table I. Published data on patients with aortoesophageal (AEF) fistula caused by esophageal stent
604.e20 Cas cliniques
NBCA is a low-viscosity liquid embolic material that is usually used for occlusion of arteriovenous malformation. NBCA embolization is a very effective and useful method for treating active arterial bleeding when coils or particle embolization cannot be performed. NBCA is difficult to handle because of its rapid polymerization and lack of radiopacity; NBCA is usually mixed with iodized oils (such as ethiodol) at ratios ranging between 1:1 and 1:4 depending on the flow rate and occlusion level to aid opacification and slow its polymerization time. Ethiodol was added to NBCA at a ratio of 1:1 for rapid polymerization. Test injections using contrast material are useful to determine the optimal volume and injection rate before injection of NBCA. Inadequate volume and incorrect injection rate into the AEF can cause unwanted regurgitation into the aorta. The catheter should be filled with a solution of 5% dextrose in water before NBCA injection because any contact with an ionic substance causes it to polymerize rapidly. After NCBA injection, further flushing of the catheter with 5% dextrose in water is necessary to prevent occlusion or adhesion of the catheter.16 Only two reports have described NBCA embolization for the AEF and aortoenteric fistula.17-19 We suggest that NBCA embolization is useful for the prompt control of bleeding in hemodynamically unstable patients. Although NBCA was displaced after initial injection, we achieved successful embolization after additional injections. We hypothesized that NBCA embolization alone was insufficient and had a temporary effect, which is supported by a previous report of reoccurrence of bleeding after NBCA embolization alone was performed for an AEF.17 However, in this case we used NBCA embolization as a bridge to stent graft placement. We were able to consider stent graft placement or surgical repair only after the patient’s hemodynamics became stable. AEF has been treated by open surgical management such as graft interposition and esophageal reconstruction after open thoracotomy.6-8 Recently, endovascular management has emerged as a lessinvasive therapeutic option for AEFs. Endovascular aortic stent graft placement is a safe and effective method of rapidly controlling acute bleeding from an AEF.9-15 However, endovascular stent graft placement is a longer procedure than NBCA embolization, because of the time required for arteriotomy of the femoral artery and placement of the stent graft itself. Massive, life-threatening bleeding from the aorta is a time-critical condition, and this case report suggests that NBCA embolization of the fistula can be performed more quickly, within
Vol. 24, No. 4, 2010
several minutes, and is particularly beneficial for patients with hypovolemic shock. Endovascular aortic stent graft placement is not recommended when clinical signs of infection are present.7,20,21 In this case, no clinical signs of infection were observed. NBCA embolization of the fistula tract may help reduce the possibility of infection or sepsis because the fistula is closed during embolization. If a fistula remains between the esophagus and mediastinum, it could lead to mediastinitis or graft infection after placement of the aortoesophageal stent. Thus, NBCA embolization of the fistula tract and subsequent endovascular stent graft placement coupled with antibiotic therapy may be a safer alternative to surgical repair. In conclusion, NBCA embolization of an AEF is a prompt treatment that can control massive bleeding in a hemodynamically unstable patient. Endovascular stent graft placement following fistula closure with NBCA is a safe method for the treatment of an AEF and may be an alternative to surgical repair. REFERENCES 1. Allgaier HP, Schwacha H, Technau K, et al. Fatal esophagoaortic fistula after placement of a self-expanding metal stent in a patient with esophageal carcinoma. N Engl J Med 1997;11(337):1778. 2. Siersema PD, Tan TG, Sutorius FF, et al. Massive hemorrhage caused by a perforating Gianturco-Z stent resulting in an aortoesophageal fistula. Endoscopy 1997;29:416-420. 3. Rogart J, Greenwald A, Rossi F, et al. Aortoesophageal fistula following Polyflex stent placement for refractory benign esophageal stricture. Endoscopy 2007;39:321-322. 4. Unosawa S, Hata M, Sezai A, et al. Surgical treatment of an aortoesophageal fistula caused by stent implantation for esophageal stenosis : report of a case. Surg Today 2008;38: 62-64. 5. Um SJ, Park BH, Son C. An aortoesophageal fistula in patient with lung cancer after chemo-irradiation and subsequent esophageal stent implantation. J Thorac Oncol 2009;4:263-265. 6. Hollander JE, Quick G. Aortoesophageal fistula: a comprehensive review of the literature. Am J Med 1991;91:279-287.
Cas cliniques 604.e21
7. Prokakis C, Koletsis E, Apostolakis E, et al. Aortoesophageal fistulas due to thoracic aorta aneurysm: surgical versus endovascular repair. Is there a role for combined aortic management? Med Sci Monit 2008;14:48-54. 8. Topel I, Stehr A, Steinbauer MG, et al. Surgical strategy in aortoesophageal fistulae: endovascular stentgrafts and in situ repair of the aorta with cryopreserved homografts. Ann Surg 2007;246:853-859. 9. Leobon B, Roux D, Mugniot A, et al. Endovascular treatment of thoracic aortic fistulas. Ann Thorac Surg 2002;74: 247-249. 10. Allen RC, Sebastian MG. The role of endovascular techniques in aortoesophageal fistula repair. J Endovasc Ther 2001;8:602-603. 11. Taylor BJ, Stewart D, West P, et al. Endovascular repair of a secondary aortoesophageal fistula : a case report and review of the literature. Ann Vasc Surg 2007;21:167-171. 12. Assink J, Vierhout BP, Snellen JP, et al. Emergency endovascular repair of an aortoesophageal fistula caused by a foreign body. J Endovasc Ther 2005;12:129-133. 13. Kato N, Tadanori H, Tanaka K, et al. Aortoesophageal fistula : relief of massive hematemesis with an endovascular stent-graft. Eur J Radiol 2000;34:63-66. 14. Flores J, Shiiya N, Kunihara T, et al. Aortoesophageal fistula: alternatives of treatment case report and literature review. Ann Thorac Cardiovasc Surg 2004;10:241-246. 15. Song Y, Liu Q, Shen H, et al. Diagnosis and management of primary aortoenteric fistulas experience learned from eighteen patients. Surgery 2008;143:43-50. 16. Pollak JS, White RI. The use of cyanoacrylate adhesives in peripheral embolization. J Vasc Interv Radiol 2001;12: 907-913. 17. Reedy FM. Embolization of aortoesophageal fistula: a new therapeutic approach. J Vasc Surg 1988;8:349-350. 18. Finch L, Heathcock RB, Quigley T, et al. Emergent treatment of a primary aortoenteric fistula with N-butyl 2-cyanoacrylate and endovascular stent. J Vasc Interv Radiol 2002;13:841-843. 19. Mok VW, Ting AC, Law S, et al. Combined endovascular stent grafting and endoscopic injection of fibrin sealant for aortoenteric fistula complicating esophagectomy. J Vasc Surg 2004;40:1234-1237. 20. Burks JA Jr, Faries PL, Gravereaux EC, et al. Endovascular repair of bleeding aortoenteric fistulas: a 5-year experience. J Vasc Surg 2001;34:1055-1059. 21. Gonzalez-Fajardo JA, Gutierrez V, Martin-Pedrosa M, et al. Endovascular repair in the presence of aortic infection. Ann Vasc Surg 2005;19:94-98.