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1 Korean J Gastroenterol Vol. 64 No. 4, pissn eissn CASE REPORT Overtube 를이용한식도정맥류결찰술후발생한식도천공과지연성합병증으로나타난종격동염의치료 김선웅, 이윤정, 김수정, 이경언, 김아란, 박상우 1, 최원혁, 심찬섭 건국대학교의과대학건국대학교병원내과학교실, 영상의학교실 1 Overtube-related Delayed Esophageal Perforation with Mediastinitis Sun Woong Kim, Yoon Jeong Lee, Soo Jung Kim, Kyung Ann Lee, Ah Ran Kim, Sang Woo Park 1, Won Hyeok Choe and Chan Sup Shim Departments of Internal Medicine and Radiology 1, Konkuk University Hospital, Konkuk University School of Medicine, Seoul, Korea Overtube provides a conduit for the passage of endoscope into the digestive tract. Esophageal perforation with mediastinitis is a rare overtube-related complication. Until now, no reports have been published regarding the esophageal perforation which developed many months after the original procedure using the overtube. A 56-year-old female visited our hospital complaining of chest pain and back pain that began 14 days ago. The patient underwent esophageal variceal ligation using the overtube 12 months earlier. She was diagnosed with esophageal perforation with mediastinitis which extended to intervertebral and epidural space. The cause of this condition was considered to have been related to the use of overtube. Management of delayed perforation remains controversial. Although surgical management might be the preferred mode of treatment, she underwent local N-butyl 2-cyanoacrylate injection therapy and temporary stent therapy with antibiotics due to high operative risk. Herein, we report a case of overtube-related delayed esophageal perforation with mediastinitis that was successfully treated by nonoperative management. (Korean J Gastroenterol 2014;64: ) Key Words: Esophageal perforation; Mediastinitis; Overtube; Stents 서론 주요상부위장관출혈원인의 30% 이상이식도정맥류출혈이다. 식도정맥류결찰술은급성식도정맥류출혈에권고되는치료법이다. 시술의부작용으로일시적인고열과연하곤란, 통증이주로나타난다. 식도천공의발생은매우드물다. 최근까지보고된증례들은시술중혹은시술후입원관찰기간동안에식도천공을진단하였다. 1,2 식도천공의원인과치료방법이다양하고, 증례의수가적고, 사망률이높은이유로수술적치료와비수술적치료의지침이정립되지않았지만, 종격동혹은흉강에광범위한조직오염이있는경우에는수술적치료를시행하는것이권고되고있다. 3,4 저자들은식도정맥류결찰술후지연되어증상이발생한광범위한주변조직의오염과급성종격동염을동반한식도천공을비수술적치료로성공적으로치료한 1예를경험하였기에문헌고찰과함께보고한다. 증례 56세여자가내원 2주전부터발생한가슴과허리통증을주소로내원하였다. 환자는과거부터만성 B형간염에의한간경변증으로라미부딘으로치료중이었으며, 당뇨로인한만성신부전으로퓨로세마이드를복용중이었다. 또한환자는간경변증에의한합병증으로내원 12개월전에식도정맥류출혈 Received March 3, Revised April 15, Accepted April 21, CC This is an open access article distributed under the terms of the Creative Commons Attribution Non-Commercial License ( by-nc/3.0) which permits unrestricted non-commercial use, distribution, and reproduction in any medium, provided the original work is properly cited. 교신저자 : 최원혁, , 서울시광진구능동로 120-1, 건국대학교병원소화기병센터 Correspondence to: Won Hyeok Choe, Digestive Disease Center, Konkuk University Hospital, Neungdong-ro, Gwangjin-gu, Seoul , Korea. Tel: , Fax: , @kuh.ac.kr Financial support: None. Conflict of interest: None. Korean J Gastroenterol, Vol. 64 No. 4, October

Kim SW, et al. Overtube-related Esophageal Perforation 225 Fig. 1. Endoscopic findings. (A) Endoscopic elastic band ligation for active variceal hemorrhage was performed 12 months ago.

(C) Follow-up endoscopic finding at admission showed esophageal fistula (arrowhead) on mid-esophagus. Fig. 2. MRI findings.

(B) Follow-up MRI taken after 4 months of treatment shows markedly decreased extent of posterior mediastinal inflammatory lesion. 이있어내경 15 mm, 장경 19.

그후약 12개월동안특별한임상증상없이지내던중가슴과허리부위에전체적으로발생한둔통으로검사를진행하였다. 환자는흡연과음주를하지않았고, 내원시혈압 131/73 mmhg, 맥박수 81회 / 분, 호흡수 20회 / 분, 체온은 37.3 o C였다. 결막은약간창백하였고공막에황달은관찰되지않았다.

7 mg/dl, AST 48 U/L, ALT 16 U/L, 총단백 8.4 g/dl, 알부민 3.0 mg/dl였다. 혈액응고검사에서프로트롬빈시간은 1.30 INR (70%) 이었다. 적혈구침강속도는 30 mm/hr, C반응단백은 4.87 mg/dl로증가소견을보였다.

2 Kim SW, et al. Overtube-related Esophageal Perforation 225 Fig. 1. Endoscopic findings. (A) Endoscopic elastic band ligation for active variceal hemorrhage was performed 12 months ago. (B) Follow-up endoscopy at 3 month after the procedure revealed small esophageal diverticulum at the ligation site. (C) Follow-up endoscopic finding at admission showed esophageal fistula (arrowhead) on mid-esophagus. Fig. 2. MRI findings. (A) Posterior mediastinal inflammatory mass that extends to intervertebral and epidural space can be seen (arrowheads). (B) Follow-up MRI taken after 4 months of treatment shows markedly decreased extent of posterior mediastinal inflammatory lesion. 이있어내경 15 mm, 장경 19.5 cm 크기의 overtube를이용한식도정맥류결찰술을 1회시행하였던병력이있었다 (Fig. 1A). 시술 3개월후에외래에서추적관찰한상부위장관내시경에서는절치에서약 20 cm 하방에위치한식도게실이외에특이소견은없었으며, 단순흉부방사선사진에서도이상소견은없었다 (Fig. 1B). 그후약 12개월동안특별한임상증상없이지내던중가슴과허리부위에전체적으로발생한둔통으로검사를진행하였다. 환자는흡연과음주를하지않았고, 내원시혈압 131/73 mmhg, 맥박수 81회 / 분, 호흡수 20회 / 분, 체온은 37.3 o C였다. 결막은약간창백하였고공막에황달은관찰되지않았다. 호흡은정상이었고, 심잡음은청진되지않았으며흉부와복부에압통은없었다. 일반혈액검사에서혈색소 15.0 g/dl, 백혈구 /mm 3, 혈소판 /mm 3 였고, 생화학검사에서혈액요소질소 21.8 mg/dl, 크레아티닌 2.00 mg/dl, 나트륨 130 meq/l, 칼륨 4.4 meq/l, 총빌리루빈 0.7 mg/dl, AST 48 U/L, ALT 16 U/L, 총단백 8.4 g/dl, 알부민 3.0 mg/dl였다. 혈액응고검사에서프로트롬빈시간은 1.30 INR (70%) 이었다. 적혈구침강속도는 30 mm/hr, C반응단백은 4.87 mg/dl로증가소견을보였다. 환자는이뇨제로복수를조절중이었던상태로중등도의간장애 (Child-Pugh class B) 를보였다. 심전도에서이상소견은없었다. 단순흉부방사선사진에서종격동기종, 기흉, 기복의소견은관찰되지않았다. 컴퓨터단층촬영과자기공명검사를시행하였으며, 검사에서식도벽이비후된소견과후부종격동에서척추간공간까지분포한경계가불분명한염증성병변이관찰되었다 (Fig. 2A). 식도천공에의한종격동염을의심하여상부위장관내시경과식도조영술을시행하였다. 이전에관찰된식도게실부위에식도누공이관찰되었으며 (Fig. 1C), 이는식도조영술에서도확인되었다. 이를치료하기위해입원하여치료적금식을실시하고항생제를정맥주사하기시작하였다. 식도천공을치료하기위해누공에미세도관을통하여시아노아크릴레이트 0.5 ml를주입치료하였다. 하지만시아노아크릴레이트주입후 2일째에시행한식도조영술에서여전히소량의조영제누출이지속되었다 (Fig. 3A). 추가적인치료를위해직경 18 mm, 길이 8 cm 크기의막형자가팽창형스텐트 (fully covered self-ex- Vol. 64 No. 4, October 2014

226 김선웅등. Overtube 를이용한식도정맥류결찰술후발생한식도천공과종격동염 Fig. 4. Chest X-ray finding. Esophageal stent is observed in the upper esophagus. Fig. 3. Constrast esophagram findings.

pandable metal stents, FCSEMS) 를삽입하였다 (Fig. 4). 환자는스텐트삽입후 C반응단백수치가감소하기시작하였으며식도조영술에서더이상누출이관찰되지않았다. 임상증상호전으로스텐트삽입후 5일째부터경구식이를시작하였고, 스텐트는 3주간유지한후제거하였다. 스텐트제거후에도식도조영술에서누출이관찰되지않았다 (Fig. 3B).

역시추적관찰한척추자기공명검사에서도염증성병변이소실되었음을확인하였다 (Fig. 2B). 이후에환자는종격동염의재발이없었으며, 간기능의변화없이지냈다. 18개월후복통과고열을동반한간경변증의합병증으로발생한자발성세균성복막염에의한패혈증및간신증후군으로사망하였다. 고 찰 식도정맥류결찰술에서 overtube는식도정맥류출혈의치료중에반복적인삽관을돕는보조적인목적으로이용된다.

3 226 김선웅등. Overtube 를이용한식도정맥류결찰술후발생한식도천공과종격동염 Fig. 4. Chest X-ray finding. Esophageal stent is observed in the upper esophagus. Fig. 3. Constrast esophagram findings. (A) Leakage of dye (arrowheads) from posterior aspect of upper esophagus is demonstrated. (B) Follow-up esophagram taken 3 days after stent removal shows no leakage of contrast agent. pandable metal stents, FCSEMS) 를삽입하였다 (Fig. 4). 환자는스텐트삽입후 C반응단백수치가감소하기시작하였으며식도조영술에서더이상누출이관찰되지않았다. 임상증상호전으로스텐트삽입후 5일째부터경구식이를시작하였고, 스텐트는 3주간유지한후제거하였다. 스텐트제거후에도식도조영술에서누출이관찰되지않았다 (Fig. 3B). 종격동염치료를위해세프트리악손과메트로니다졸을 6주간투약하였으며, 가슴과허리부위의둔통소실과 C단백반응의정상화 (0.90 mg/dl) 및자기공명검사에서종격동염이치유되었음을확인할수있었다. 치료후 3개월간격으로추적관찰한식도조영술에서누출은관찰되지않았으며, 시술 4개월후에시행한내시경검사에서도반흔만관찰되었을뿐더이상의누출을시사하는소견은관찰되지않았다. 역시추적관찰한척추자기공명검사에서도염증성병변이소실되었음을확인하였다 (Fig. 2B). 이후에환자는종격동염의재발이없었으며, 간기능의변화없이지냈다. 18개월후복통과고열을동반한간경변증의합병증으로발생한자발성세균성복막염에의한패혈증및간신증후군으로사망하였다. 고 찰 식도정맥류결찰술에서 overtube는식도정맥류출혈의치료중에반복적인삽관을돕는보조적인목적으로이용된다. Multiband ligator의발명이후로식도정맥류결찰술에서사 용의빈도는감소하였지만, 식도내이물질의제거, 소장내시경등다양한시술의보조적인목적으로임상에서자주이용되고있다. Overtube로인한합병증으로식도점막의손상, 식도천공, 정맥류의파열, 피열연골의손상, 그리고췌장염등이보고되었다. 1992년 Goldschmiedt 등 2 이최초로식도천공이발생한증례를보고하였고, 이후로식도천공이발생한증례가최소한 7개이상보고되었다. 5 보고된증례는모두시술후수일이내에식도천공을진단하였다. 1997년 Dinning 등 6 이식도정맥류결찰술시행 2주내에진단한식도천공의증례가가장지연되어증상이발생한경우였다. 저자들이보고한증례의환자는식도정맥류결찰술이후 12개월동안무증상으로일상생활을지속하였다. 식도정맥류결찰술을시행하고 12개월후지연성종격동염과식도천공이진단된증례는저자들의보고가최초이다. 식도천공은전형적으로지속적인가슴통증, 등통증, 고열, 피하기종또는종격동기종을동반한다. 7 식도천공이의심되는환자에서우선단순흉부방사선사진을촬영한다. 식도천공환자의 42% 에서종격동기종소견을보이며, 기흉또는기복의소견도각각 30% 의환자에서관찰할수있다. 8 생체징후가안정된환자의경우에는확진을위하여식도조영술을시행한다. 3 상부위장관내시경은즉각적으로병변의확인이가능하고민감도가매우높지만, 시술중주입된공기가병변의악화나폐기종을유발할가능성이있다. 컴퓨터단층촬영검사는식도조영술시행이불가능하거나, 식도천공의위치를찾기어려운경우에유용하다. 이번증례의경우, 식도정맥류결찰술 3개월후에시행한추적상부위장관내시경에서천공이관찰되지않았다. 내시경검사의높은민감도에불구하고, 식도게 The Korean Journal of Gastroenterology

4 Kim SW, et al. Overtube-related Esophageal Perforation 227 실부위에발생한식도천공을확인하지못하였을가능성도있다. 추적내시경검사를시행한시기에전형적인임상증상이없었고, 단순흉부방사선사진이정상소견이었으므로, 검사자가식도천공을의심하지않은상태에서식도게실부위를면밀하게확인할만큼의충분한공기주입없이추적내시경검사를종료하였을가능성이있기때문이다. 따라서식도게실을동반한환자에서 overtube를이용한치료내시경시술을하는경우에 overtube로인한합병증에대한주의와추적검사에서의면밀한관찰이필요하다. 식도천공의치료에는비수술적치료와수술적치료가있다. 환자의전신상태, 천공의크기와위치, 누출된오염물이침범한범위, 동반질환을고려하여치료의방법을선택할수있다. 패혈증이동반되었거나, 천공이크고, 주변조직의광범위한오염이관찰되는경우에는수술적치료를권고하고있다. 3,4 수술적치료에는단순봉합술과식도절제술, 배액관삽입술, T-tube 삽입술이있으며, 천공의위치, 식도질환에따라서치료방법을결정한다. 9 비수술적치료에는조직유착물질의주입술, 스텐트삽입술, 내시경클립술이있다. 비수술적치료는공통적으로최소 48시간이상의금식과 7일에서 14일동안의광범위항생제투약을포함한다. 이번증례의환자는후부종격동에서척추간공간까지광범위하게오염된병변을보여수술적치료에적합한대상이었다. 하지만환자는 B형간염에의한간경변증과만성신부전증을동반하고있어수술적치료의고위험군에해당하였기때문에, 저자들은비수술적치료를우선적으로시도하였다. 시아노아크릴레이트는체액과접촉하면중합체를형성하는조직유착물질로개구부의크기가작은누공의치료에적합하다. 즉각적인반응속도를늦추기위해서리피오돌과 1:1에서 1:1.5의비율로희석하여혼합액 1 ml를개구부에주입하며, 크기가 1 cm인식도기관지누공에서좋은결과를보인보고가있었다. 10 이번증례의환자역시 1 cm 미만의개구부를보였기때문에, 저자들은누공의개구부에시아노아크릴레이트를주입하는치료를시행하였다. 하지만시술 2일후에시행한식도조영술검사에서누출이지속되는결과를보였다. 식도천공에서스텐트삽입술은안전하고효과적인치료방법으로점차사용이증가하고있다. 패혈증을동반하지않은환자에서천공의크기가작고, 상부식도조임근, 하부식도조임근과충분한간격이있는경우에스텐트삽입술의시행이권고된다. 11 보편적으로사용하는스텐트에는 FCSEMS와자가팽창형막부착형플라스틱스텐트 (self-expanding plastic stents, SEPS) 가있으며치료효과는큰차이가없다. 12 SEPS 는가격이저렴하고, 표면이부드러워쉽게제거할수있는반면에, FCSEMS와비교하여스텐트의이탈이더욱빈번하 게발생한다는단점이있다. 13 FCSEMS의경우에도 10-35% 에서이탈이발생하는것으로보고되어있으나, Niti-S 스텐트 (Taewoong Medical, Seoul, Korea) 는 7% 에서만이탈이발생하였다. 14 또한스텐트삽입술의합병증으로육아조직의증식이빈번하게발생하기때문에, 누출이없는것을확인한시점으로부터 2주에서 6주후에는스텐트를제거해야한다. 11,15 증례의환자는생체지수가안정되었고, 누공의위치와크기가스텐트삽입술의대상으로적합하였다. Niti-S 스텐트를삽입한후에시행한식도조영술에서누출이관찰되지않았고, 3주후스텐트를제거한이후에도누출이관찰되지않는좋은결과를보였다. 저자들은 overtube를이용한식도정맥류결찰술 12개월후진단된종격동염을동반한식도천공에대하여비수술적치료시술로치료한 1예를경험하였기에문헌고찰과함께보고한다. REFERENCES 1. Schmitz RJ, Sharma P, Badr AS, Qamar MT, Weston AP. Incidence and management of esophageal stricture formation, ulcer bleeding, perforation, and massive hematoma formation from sclerotherapy versus band ligation. Am J Gastroenterol 2001; 96: Goldschmiedt M, Haber G, Kandel G, Kortan P, Marcon N. A safety maneuver for placing overtubes during endoscopic variceal ligation. Gastrointest Endosc 1992;38: Lindenmann J, Matzi V, Neuboeck N, et al. Management of esophageal perforation in 120 consecutive patients: clinical impact of a structured treatment algorithm. J Gastrointest Surg 2013;17: Carrott PW Jr, Low DE. Advances in the management of esophageal perforation. Thorac Surg Clin 2011;21: Wells CD, Fleischer DE. Overtubes in gastrointestinal endoscopy. Am J Gastroenterol 2008;103: Dinning JP, Jaffe PE. Delayed presentation of esophageal perforation as a result of overtube placement. J Clin Gastroenterol 1997;24: Michel L, Grillo HC, Malt RA. Operative and nonoperative management of esophageal perforations. Ann Surg 1981;194: Hasan S, Jilaihawi AN, Prakash D. Conservative management of iatrogenic oesophageal perforations--a viable option. Eur J Cardiothorac Surg 2005;28: Brinster CJ, Singhal S, Lee L, Marshall MB, Kaiser LR, Kucharczuk JC. Evolving options in the management of esophageal perforation. Ann Thorac Surg 2004;77: Kim HG, Cho JW, Park SJ, et al. Two cases of alimentary tract fistula treated by endoscopic local injection therapy. Korean J Gastrointest Endosc 2003;26: van Heel NC, Haringsma J, Spaander MC, Bruno MJ, Kuipers EJ. Short-term esophageal stenting in the management of benign Vol. 64 No. 4, October 2014

5 228 김선웅등. Overtube 를이용한식도정맥류결찰술후발생한식도천공과종격동염 perforations. Am J Gastroenterol 2010;105: Radecke K, Gerken G, Treichel U. Impact of a self-expanding, plastic esophageal stent on various esophageal stenoses, fistulas, and leakages: a single-center experience in 39 patients. Gastrointest Endosc 2005;61: Langer FB, Wenzl E, Prager G, et al. Management of postoperative esophageal leaks with the Polyflex self-expanding covered plastic stent. Ann Thorac Surg 2005;79: Shim CS. Esophageal stent for cervical esophagus and esophagogastric junction. Clin Endosc 2012;45: El Hajj II, Imperiale TF, Rex DK, et al. Treatment of esophageal leaks, fistulae, and perforations with temporary stents: evaluation of efficacy, adverse events, and factors associated with successful outcomes. Gastrointest Endosc 2014;79: The Korean Journal of Gastroenterology

노영남

노영남 Purpose: Delayed massive hemorrhages from pseudoaneurysm rupture of the peripancreatic large arteries, after pancreaticoduodenectomy, are fatal. We reviewed the clinical course and outcome of bleeding