Aortoesophageal Fistula after Thoracic Endovascular Aortic Repair Diagnosed and Followed with Endoscopy

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1 CASE REPORT Aortoesophgel Fistul fter Thorcic Endovsculr Aortic Repir Dignosed nd Followed with Endoscopy Disuke Chi 1,2, Norihiro Hnt 1, Ysumitsu Arki 1, Mnu Swy 1, Tetsuro Yoshimur 1, Mshiko Aoki 3, Tdshi Shimoym 1 nd Shinsku Fukud 1 Astrct A 70-yer-old mn reported dysphgi two months fter undergoing thorcic endovsculr ortic repir (TEVAR). An endoscopic exmintion reveled fistul etween the esophgus nd the thorcic ortic neurysm, nd computed tomogrphy (CT) showed tht the thorcic ortic neurysm hd incresed in size. The ptient ws dignosed with n ortoesophgel fistul (AEF), nd surgicl replcement of the thorcic ort ws performed. AEFs re rre ut typiclly ftl compliction fter TEVAR. Physicins should consider dignosis of AEF nd perform endoscopic exmintions nd CT in ptients who undergo TEVAR nd susequently complin of dysphgi. Key words: ortoesophgel fistul (AEF), thorcic endovsculr ortic repir (TEVAR), endoscopy () () Introduction Aortoesophgel fistuls (AEF) re rre disorder tht cn occur s n unusul compliction fter thorcic endovsculr ortic repir (TEVAR). Worldwide, TEVAR hs ecome stndrd tretment for ortic neurysms nd dissections ecuse it is ssocited with lower rtes of postopertive moridity thn conventionl open surgery. There is no estlished tretment for AEFs, nd the mortlity rte ssocited with the disorder is high. Performing endoscopic exmintions is helpful in mking prompt nd definite dignosis of AEF. However, there hve een few cse reports in which endoscopic follow-up ws performed fter surgery for AEF. We herein present cse of AEF tht developed fter TEVAR in which the ptient ws dignosed on n endoscopic exmintion nd computed tomogrphy (CT) nd treted with thorcotomy. Cse Report A 70-yer-old mn who hd undergone TEVAR in Decemer 2009 reported dysphgi two months fter the procedure. CT showed tht the neurysm of the thorcic ort hd incresed fter TEVAR, thus forming flse lumen nd compressing the esophgus (Fig. 1). The ptient ws dmitted to the deprtment of crdiovsculr surgery t Hiroski University Hospitl. The mn ws hevy smoker (40 cigrettes dy for 50 yers). A lood test reveled n elevted level of C-rective protein (6.2 mg/dl) (Tle). An endoscopic exmintion performed fter dmission reveled protrusion (similr to sumucosl tumor) with n ssocited defect of the esophgel wll tht exposed the cogul nd necrotic tissue t the upper section of the esophgus (20 cm from the incisor) (Fig. 2). Even thin scope (Olympus GIF-XP260) could not pss through the portion due to stenosis. Susequent ngiogrphy reveled n extrvstion from the proximl side of the stent plced t the descending thorcic ort (Fig. 3). The ptient ws dignosed s hving n AEF, nd removl of the stent-grft, replcement of the descending ort with prosthetic grft nd omentl implnttion were performed. A proton-pump inhiitor (PPI) ws dministered during the therpeutic term. Two weeks fter surgery, endoscopic exmintions showed reduction in the size of the cogul nd necrotic tissue (Fig. 4), nd further reductions with regenerting Deprtment of Gstroenterology, Hiroski University Grdute School of Medicine, Jpn, Deprtment of Internl Medicine, Kensei Hospitl, Jpn nd Deprtment of Rdiology, Hiroski University Grdute School of Medicine, Jpn Received for puliction Octoer 16, 2012; Accepted for puliction Novemer 19, 2012 Correspondence to Dr. Tdshi Shimoym, tsimo-hki@umin.c.jp 451

2 mucos were oserved four weeks fter surgery (Fig. 4). The ptient showed no complictions fter tking liquid food nd ws dischrged on the 79th dy postsurgery. Five Tle. Lortory Dt on Admission Peripherl lood WBC 7,560 Neutro 66.3 Bso 0.7 Eosino 2.1 Lympho 23.9 Mono 7.0 RBC H 14.9 Ht 44.4 Plt Blood chemistory TP 8.2 g/dl Al 4.3 g/dl T-il 1.3 mg/dl AST 18 IU/L ALT 12 IU/L LDH 159 IU/L ALP 222 IU/L Ȗ-GTP 159 IU/L AMY 62 IU/L BUN 20 mg/dl Cr 1.0 mg/dl CRP 6.2 mg/dl WBC: white lood cell, RBC: red lood cell, H: hemogloin, Ht: hemtocrit, Plt: pltelets, Al: lumin, T-il: totl iliruin, AST: sprtte minotrnsferse, ALT: lnine minotrnsferse, LDH: lctte dehydrogense, ALP: lkline phosphtse, Ȗ-GTP: Ȗ-glutmyl trnspeptidse, AMY: mylse, BUN: lood ure nitrogen, Cr: cretinine, CRP: C-rective protein g/dl months fter surgery, CT confirmed the reduction in size of the thorcic ortic neurysm (Fig. 5), nd n endoscopic exmintion showed tht the rownish irremovle necrotic mss ws still present in the esophgus (Fig. 4c). CT performed 10 months fter surgery reveled the lmost complete disppernce of the thorcic neurysm (Fig. 5). The ptient remins well without ny significnt complictions, nd ttends our hospitl for regulr exmintions. Discussion Recently, the numer of ptients who undergo TEVAR for thorcic ortic disese hs incresed due to the minimlly invsive nture of this technique in comprison with open repir (1). However, TEVAR is ssocited with severl complictions, including prplegi, stroke, post-implnttion syndrome, device migrtion nd AEF formtion (2). AEF formtion is the most lethl of these complictions. Indeed, in n investigtion of the previous literture, Murdi et l. found tht only six of 24 ptients (25) who developed AEFs fter undergoing TEVAR could e rescued (3). Egge- c d Figure 1. CT imges showing the thorcic ortic neurysm efore TEVAR () nd the incresed neurysm two months fter TEVAR (). A coronl section (c) nd 3D thorcic ortic imge (d) were constructed. 452

3 Figure 2. Endoscopic findings showed protrusion similr to sumucosl tumor with ulcertion t the posterior wll of the upper section of the esophgus. Figure 3. Angiogrphy reveled n extrvstion from the proximl side of the stent plced t the descending thorcic ort (rrow). c Figure 4. Endoscopic exmintions were performed two weeks (), four weeks () nd five months (c) fter surgery. The scope (GIF-XP260) ws le to e pssed through the stenotic prt of the esophgus four weeks fter the surgery. recht et l. lso reported tht six of 268 ptients who hd undergone TEVAR developed AEFs (incidence 1.9) nd tht ll of these ptients died due to ftl leeding or medistinitis (4). Mechnicl dmge cused y rteril pulstion, stentgrft rigidity nd interruption of the circultion of the esophgel wll y enlrged neurysms re ll expected to ply roles in the development of AEFs (4). In our cse, flse lumen of the thorcic ortic neurysm grdully formed fter TEVAR due to lood flow entry etween the stent nd the neurysm (perigrft lek). As result, the esophgel wll ws compressed y the neurysm. The clinicl symptoms of AEFs generlly consist of Chiri s trid: midthorcic chest pin, sentinel rteril hemorrhge 453

4 Figure 5. CT performed t five months fter surgery showed tht the thorcic ortic neurysm hd reduced in size (). CT performed 10 months fter surgery showed tht the thorcic neurysm hd lmost completely disppered (). nd ftl hemorrhge (5). The most common symptom of AEFs is hemtemesis (6), nd the presenttion of only dysphgi in the sence of the trid, s seen in our ptient, is rre. There hve een severl reports tht involved the exmintion of AEFs using endoscopy (4, 7-10). The most common form of AEF is sumucosl tumor-like protrusion; other forms include extrinsic compression, ulcertive lesions, leeding only, pulsting protrusions with centrl fistultion nd exposure of the ortic stent-grft. The endoscopic findings oserved in our cse consisted of protrusion similr to sumucosl tumor, exposing the cogul nd necrotic tissue. In most previous reports, the AEFs were locted etween the posterior to the left lterl wll of the esophgus. The loction of the lesion my e useful for mking dignosis of AEF. There is no literture reporting endoscopic findings fter surgery for AEF. In the present cse, omentl implnttion ws performed to void esophgectomy; thus, the defect of the esophgel wll with cogul nd necrotic tissue persisted for more thn five months. However, the dysphgi disppered nd did not recur fter surgery. In this cse, we performed smll clier endoscopy due to the presence of esophgel stenosis. However, the lod of the endoscopic exmintions my constitute risk for rupture of the ortic neurysm. Trnsnsl smll clier endoscopy is sfer procedure thn trnsorl conventionl endoscopy prticulrly in terms of crdiopulmonry effects (11, 12). Therefore, for the mngement of ptients suspected of hving AEFs, trnsnsl endoscopy is considered fter CT. In conclusion, the development of AEFs fter TEVAR is rre ut typiclly ftl compliction. We were le to dignose the ptient with n AEF nd successfully tret the condition, lthough the ptient presented with dysphgionly, without hemtemesis. Physicins should therefore consider dignosis of AEF nd perform CT first followed y endoscopic exmintions in ny ptients who hs undergone TEVAR nd complins of dysphgi. The uthors stte tht they hve no Conflict of Interest (COI). References 1. Wlsh SR, Tng TY, Sdt U, et l. Endovsculr stenting versus open surgery for thorcic ortic disese: systemtic review nd met-nlysis of periopertive results. J Vsc Surg 47: , Grce JW, Ronld MF. Endovsculr repir of the Thorcic ort. Semin Intervent Rdiol 26: 17-24, Murdi A, Ymguchi M, Kitgw A, et l. Secondry Aortoesophgel fistul fter thorcic endovsculr ortic repir for huge neurysm. Dign Interv Rdiol 2012, DOI: / Eggerecht H, Meht RH, Dechene A, et l. Aortoesophgel fistul fter thorcic ortic stent-grft plcement: rre ut ctstrophic compliction of novel emerging technique. JACC Crdiovsc Interv 2: , Chiri H. Fremdkorpeverletzung des Oesophgus mit Aortenperfortion. Ber Klin Wochenschr 51: 7-9, 1914 (in Germn). 6. Ygi N, Akiym H, Igki N, et l. Two cses of ortogstrointestinl fistul. Intern Med 38: , Sog K, Kitmur R, Tkenk S, et l. Progressive endoscopic findings in cse of ortoesophgel fistul. Dig Endosc 24: 210, Unosw S, Akiym K, Nkt K, et l. Successful surgicl tretment for n ortoesophgel fistul due to descending ortic neurysm. Ann Thorc Criovsc Surg 9: , Gvens E, Zidi Z, AI-Jundi W, et l. Aortoesophgel fistul fter endovsculr ortic neurysm repir of mycotic neurysm. Int J Vsc Med 2011: , Morishim K, Hosoy Y, Ari W, et l. Remnnt descencing ort fter surgery for dissecting neurysm of the thorcic ort: delyed perfortion of the esophgus due to ruputured ortic neurysm. Endoscopi Digestiv 15: , 2003 (in Jpnese). 11. Yuki M, Amno Y, Komzw Y, et l. Unsedted trnsnsl 454

5 smll-clier esophgogstroduodenoscopy in elderly nd edridden ptients. World J Gstroenterol 15: , Kwi T, Ymmoto K, Hukuzw M, et l. Ultr-thin trnsnsl esophgogstroduodenoscopy. Nippon Rinsho 7: , 2010 (in Jpnese) The Jpnese Society of Internl Medicine 455