CASE REPORTS THE KOREAN JOURNAL OF PANCREAS AND BILIARY TRACT 금속스텐트협착이발생한간문부담관암에서내시경적풍선확장술시행후발생한간동맥가성동맥류 1예 김선민, 장슬아, 윤지영, 김유지, 정주영, 송수경, 김성훈, 이승옥 전북대

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1 CASE REPORTS THE KOREAN JOURNAL OF PANCREAS AND BILIARY TRACT 금속스텐트협착이발생한간문부담관암에서내시경적풍선확장술시행후발생한간동맥가성동맥류 1예 김선민, 장슬아, 윤지영, 김유지, 정주영, 송수경, 김성훈, 이승옥 전북대학교의학전문대학원내과학교실 Hepatic Artery Pseudoaneurysm after Endoscopic Balloon Dilatation in Stenosis of Metallic Stent for Klatskin Tumor Seon Min Kim, Seol A Jang, Ji Young Yoon, Yu Ji Kim, Joo Young Chung, Soo Kyeong Song, Seong Hun Kim, Seung-Ok Lee Department of Internal Medicine, Chonbuk National University Medical School and Hospital, Jeonju, Korea Self-expandable metallic stent (SEMS) has been widely used for internal drainage of malignant biliary stricture. SEMS placement was considered relatively safe. However, complications, such as cholangitis, pancreatitis, bleeding, perforation, and stent migration, were occasionally observed. Especially hepatic artery pseudoaneurysm after endoscopic balloon dilatation in stenosis of SEMS had not been reported. We experienced the case that patient of hepatic artery pseudoaneurysm after endoscopic balloon dilatation in stenosis of metallic stent for Klatskin tumor and was treated by transcatheter arterial embolization. Therefore, we report this case with literature review. key words: self expandable metallic stent, balloon dilatation, pseudoaneurysm 서론 1) 수술적절제를할수없는간문부담관암환자에서자가팽창성금속스텐트 (self-expandable metal stent, SEMS) 를이용한담관배액술은담즙정체로인해발생하는질환을호전시키고, 환자의삶의질을높일수있어흔히사용되는고식적치료이다. 1 담관내 SEMS 의삽입술은비교적안전하지만담관염, 췌장염, 출혈, 천공, 스텐트이 동과같은합병증이발생할수있고, 드물게혈액담즙증의원인이될수있는간동맥가성동맥류의발생이일부보고된바있다. 2-4 하지만간문부담관암에서이미삽입된 SEMS 의협착에대해내시경적풍선확장술시행후혈액담즙증을유발시킨간동맥의가성동맥류발생이보고된증례는아직국내외에서없었다. 이제저자등은금속스텐트재협착이발생한간문부담관암에서내시경적풍선확장술시행후발생한간동맥가성동맥류 1예를경험하였기에문헌고찰과함께보고하는바이다. Corresponding author. Seung Hun Kim Department of Internal Medicine, Chonbuk National University Medical School and Hospital 567, Baekjie-daero, deokjin-gu, Jeonju-si, Jeollabuk-do, , Korea. Tel: Fax: shkimgi@jbnu.ac.kr 증례 75세여자가내원 4일전부터발생한흑색변을주소로내원하였다. 환자는 8개월전간문부담관암 (Klatskin tumor, Bismuth type II) 을진단받고수술은거부하여좌 51

김선민외 7 인 52 A B C Fig. 1. (A) There is no radiocontrast passage to the left intrahepatic bile duct due to tumor regrowth on endoscopic retrograde cholangiopancreatography (arrow).

우측간내담관및총담관에걸쳐 Y자모양으로 2개 ( 좌측길이 8 cm, 우측길이 10 cm) 의 SEMS(BONA STENT M-Hilar, Standard Sci Tech Inc., Seoul, South Korea) 삽입술을시행받았다.

0 g/dl, 혈소판 306,000/μL 였고, 혈청화학검사상 AST 924 IU/L, ALT 359 IU/L, 총빌리루빈 1.67 mg/dl, 직접빌리루빈 1.36 mg/dl, ALP 634 IU/L, γ-gtp 356 IU/L, 프로트롬빈시간 13.3초로담관폐쇄에의한간기능저하를시사하는결과가관찰되었다.

2 김선민외 7 인 52 A B C Fig. 1. (A) There is no radiocontrast passage to the left intrahepatic bile duct due to tumor regrowth on endoscopic retrograde cholangiopancreatography (arrow). (B) Endoscopic retrograde cholangiopancreatography shows dilatation of stricture in left biliary stent by balloon dilatation. (C) Endoscopic view shows hemobilia through the papilla. 우측간내담관및총담관에걸쳐 Y자모양으로 2개 ( 좌측길이 8 cm, 우측길이 10 cm) 의 SEMS(BONA STENT M-Hilar, Standard Sci Tech Inc., Seoul, South Korea) 삽입술을시행받았다. 내원 2주전 (SEMS 삽입후 7개월후 ) 환자는복통을주소로내원하였고, 이학적검사상혈압은 130/80 mmhg, 맥박은 80회 / 분, 호흡수는 20회 / 분, 체온은 38.0 o C이었다. 의식은명료하였으나급성병색을보였고우상복부의직접압통소견이관찰되었다. 검사실소견상말초혈액검사에서는백혈구 12,028/μL( 호중구 83.3%), 혈색소 9.0 g/dl, 혈소판 306,000/μL 였고, 혈청화학검사상 AST 924 IU/L, ALT 359 IU/L, 총빌리루빈 1.67 mg/dl, 직접빌리루빈 1.36 mg/dl, ALP 634 IU/L, γ-gtp 356 IU/L, 프로트롬빈시간 13.3초로담관폐쇄에의한간기능저하를시사하는결과가관찰되었다. 또한혈청 lipase 25 IU/L, amylase 16 IU/L 로췌장염은없었으나 C-반응단백은 83.4 mg/l로증가되어있었다. 복부전산화단층촬영에서종양성장에의해 SEMS 폐색소견을동반한담관염이의심되어내시경적역행성췌담관조영술 (endoscopic retrograde cholangiopancreatography, ERCP) 을시행하였고좌측간내담관에삽입된 SEMS의폐색이관찰되었다. 이미 2개의 SEMS가삽입되어있어추가스텐트삽입이어렵고비용적인부분을고려하여좌측담도에 인치유도선 (Hydra Jagwire, Boston Scientific, Natick, Mass) 을삽입한후풍선 (10 mm Hurricane balloon dilator, Boston Scientific) 을이용하여 1분씩 2회, 10 mm 내시경적풍선확장술을시행하였다. 시행후혈액담즙증이보여내시경적경비담즙배액 (endoscopic retrograde nasobiliary drainage, 이하 ENBD) 을시행하였다.(Fig. 1) 시술직후환자의혈압은 120/70 mmhg, 맥박은 90회 / 분이었고, 혈색소는 8.6 g/dl 이였으나, 다음날혈압은 110/65 mmhg, 맥박은 75 회 / 분, 혈색소 7.8 g/dl 였고, ENBD 를통해출혈을보여농축적혈구 (packed red cell, PRC) 2 pints 수혈후혈색소가 10.0 g/dl 로상승된후유지되었고보존적치료를하였다. ERCP 하풍선확장술 4일째환자는새롭게혈변을보았고, 혈압이 110/60 mmhg, 맥박은 80 회 / 분, 혈색소는 8.1 g/dl로감소되어혈액담즙증이의심되어혈관조영술을시행하였지만명확한출혈원인병소를찾지못하여종양공급혈관으로생각되는 4번간엽부위의좌측간동맥분지에 spongastan(johnson & Johnson, Soeborg, Denmark) 을이용하여예방적색전술을시행하였다.(Fig. 2) 이후환자는더이상의출혈소견보이지않아 ENBD 제거후퇴원하였다. 퇴원 1주후흑색변을주소로다시응급실로내원하였다. 이학적검사상혈압은 80/60 mmhg, 맥박은 90회 / 분, 호흡수는 20회 / 분, 체온은 38.0 o C이었다. 의식은명료하

hepatic artery (arrow). 였으나급성병색을보였고우상복부의직접압통소견이 관찰되었다. 검사실소견상말초혈액검사에서는백혈구 6,390/μL( 호중구 72.0%), 혈색소 7.

01 mg/dl, ALP 1,485 IU/L, γ-gtp 610 IU/L, 프로트롬빈시간 12.1초로출혈및담관폐쇄에의한간기능저하를시사하는결과가관찰되었다.

3) 혈색소의감소, 총빌리루빈의증가, 복부전산화단층촬영소견을종합하여우측간동맥의가성동맥류에서기인한혈액담즙증에의한출혈을의심하여혈관조영술을시행하였다.

mm 크기의가성동맥류가관찰되어, 미세도관을이용하여초선택 (superselection) 한후 4개의섬유코일 (Tornado, Cook, USA) 을이용하여색전술을시행하였고,

3 내시경적풍선확장술후발생한간동맥가성동맥류 1 예 53 Fig. 2. Angiography shows hypervascular staining tumor mass supplied by tumor feeding artery from segment 4 of left hepatic artery (arrow). 였으나급성병색을보였고우상복부의직접압통소견이 관찰되었다. 검사실소견상말초혈액검사에서는백혈구 6,390/μL( 호중구 72.0%), 혈색소 7.1 g/dl, 혈소판 249,000/ μl 였고, 혈청화학검사상 AST 641 IU/L, ALT 269 IU/L, 총빌리루빈 6.27 mg/dl, 직접빌리루빈 5.01 mg/dl, ALP 1,485 IU/L, γ-gtp 610 IU/L, 프로트롬빈시간 12.1초로출혈및담관폐쇄에의한간기능저하를시사하는결과가관찰되었다. 출혈원인을찾기위해상부위장관내시경을시행하였으나상부위장관에명확한출혈소견은관찰되지않았다. 복부전산화단층촬영에서우측간동맥에이전에보이지않았던약 mm 크기의가성동맥류가관찰되었다.(Fig. 3) 혈색소의감소, 총빌리루빈의증가, 복부전산화단층촬영소견을종합하여우측간동맥의가성동맥류에서기인한혈액담즙증에의한출혈을의심하여혈관조영술을시행하였다. 우측대퇴동맥내에 6 Fr sheath 를삽입하였고, Yashiro catheter(terumo, Tokyo, Japan) 를이용하여복강동맥과간동맥에거치시킨후촬영한영상에서우측간동맥근위부에약 7 12 mm 크기의가성동맥류가관찰되어, 미세도관을이용하여초선택 (superselection) 한후 4개의섬유코일 (Tornado, Cook, USA) 을이용하여색전술을시행하였고, 시행직후촬영한혈관조영술상에서 A B Fig. 3. At 16-day after balloon dilatation, (A) abdominal CT shows a pseudoaneurysm of right hepatic artery on transverse view, (B) Coronal view. A B Fig. 4. (A) Angiography shows a pseudoaneurysm of right hepatic artery. (B) Micro-coil was seen after embolization.

4 김선민외 7 인 54 가성동맥류가사라진것 (devascularization) 을확인할수있었다.(Fig. 4) 혈액담즙증으로인해담관의폐색이발생한것으로판단하고가성동맥류로인해추가적으로간문부를자극하는것이도움이되지않는다고판단하여좌측간내담도에경피적배액술 (percutaneous transhepatic biliary drainage, 이하 PTBD) 을동시에시행하였다. 이후환자는더이상의출혈소견보이지않았고전신상태호전되어 PTBD 를유지한상태로퇴원하였고현재 3개월째추가출혈없이외래에서추적관찰중이다. 고찰 간동맥가성동맥류는드물게발생하는질환으로, 혈관벽의손상에의해혈관이부분적으로팽창된병변을말한다. 발생원인으로는외상, 수술, 경피적경간시술 (percutaneous transhepatic procedure), 간이식, 췌장염, 담관염등이있는것으로알려져있다. 5-8 이전보고에따르면외상이나수술이나시술등의손상에의해직접적으로혈관벽에손상이발생하였거나, 담관주위의염증으로간동맥의막이약해지거나, 여러원인에의한담관내압력상승으로담관이확장되어간동맥과마찰로인한혈관벽손상에의해간동맥가성동맥류가발생할것으로생각되고있다. 2-8 간동맥가성동맥류는증상이없는것부터치명적일수있는동맥류파열까지다양하다. 약 60% 정도에서는증상없이우연히발견되는걸로알려져있다. 9 간동맥가성동맥류가파열되었을경우약 64% 에서혈액담즙증을, 약 30% 에서토혈을, 약 14% 에서혈변을보이는것으로알려져있다. 간동맥가성동맥류파열되었을경우사망률은약 21-43% 로높다. 11,12 따라서동맥류가파열되기전에진단을하는것이중요하다. 간동맥가성동맥류진단에있어혈관조영술이가장민감한검사로알려져있다. 13 동맥류가파열되었을경우보존적인치료로는지혈에실패할가능성이높으며, 색전물질을이용하여색전술을시행하는방법이가장널리이용되고있고, 효과적인치료방법이될수있다. 14,15 담도악성폐색의고식적치료로서 SEMS 삽입술시간동맥가성동맥류의발생보고는매우드물다. Rai 등은비호지킨림프종에의한담도폐색에대한고식적치료로경피적경간 SEMS 삽입술후재발된담도폐색에대해플라스틱스텐트를추가삽입한후 6개월뒤간동맥가성동맥 류가파열된증례를보고한바있다. 2 본증례는이미삽입된 SEMS내종양의성장으로인해재협착이발생한간문부담관암에서내시경적풍선확장술시행후우측간동맥에가성동맥류가발생되어출혈이발생하였고혈관색전술을이용하여치료한예로기존의보고와는달리재협착으로발견당시에는가성동맥류가없었으나내시경적풍선확장술이후발행한점이다른점이다. 본증례에서가성동맥류의발생기전을생각해보면담관주위의염증으로간동맥의막이약해졌고, 종양성장으로인한스텐트의부분폐색, 찌꺼기 (sludge) 로인한담도폐색으로인해담관내압력이상승하여담관이확장되었고, 여기에풍선확장술까지더해져담관확장이더욱확장되고마찰이일어나간동맥에미란 (erosion) 이발생하였고, 이것이더욱악화되어간동맥가성동맥류가발생되었을가능성이높아보인다. 결론적으로간문부담관암환자에서고식적인치료로흔히사용되는 SEMS 삽입후간동맥의가성동맥류와같은합병증이발생가능함을기억하는것이필요하겠고, 재협착이발생한 SEMS 내내시경적풍선확장술을고려할경우시행전복부전산화단층촬영사진에서간동맥의주행을확인하는것이필요하고, 풍선확장술시는혈관손상을주의해야하며, 시술후에도혈액담즙증, 혈변, 검은변등의출혈소견을보일경우간동맥가성동맥류발생을염두해야한다고판단된다. 요약 수술적절제를할수없는간문부담관암환자에서자가팽창성금속스텐트 (SEMS) 는비교적안전하게시행될수있으나담관염, 췌장염, 출혈, 천공, 스텐트이동과같은합병증이발생할수있고, 드물게혈액담즙증의원인이될수있는간동맥가성동맥류의발생할수있다. 하지만간문부담관암에서이미삽입된 SEMS의재협착에대해내시경적풍선확장술시행후혈액담즙증을유발시킨간동맥의가성동맥류발생이보고된증례는아직국내외에서없었다. 이제저자등은금속스텐트재협착이발생한간문부담관암에서내시경적풍선확장술시행후간동맥에가성동맥류가발생하여이를혈관색전술로치료한 1예를경험하였기에보고하는바이다.

5 내시경적풍선확장술후발생한간동맥가성동맥류 1 예 55 색인단어 : 자가팽창성금속스텐트삽입술, 풍선확장 술, 간동맥가성동맥류 참고문헌 1. Adam A, Chetty N, Roddie M, Yeung E, Benjamin I. Self-expandable stainless steel endoprostheses for treatment of malignant bile duct obstruction. American Journal of Roentgenology 1991;156: Rai R, Rose J, Manas D. Potentially fatal haemobilia due to inappropriate use of an expanding biliary stent. World Journal of Gastroenterology 2003;9: Watanabe M, Shiozawa K, Mimura T, et al. Hepatic artery pseudoaneurysm after endoscopic biliary stenting for bile duct cancer. World J Radiol 2012;4: Jeong HS ST, Hwang JC, Bae JI, Kim CW. Interventional Management of Delayed and Massive Hemobilia due to Arterial Erosion by Metallic Biliary Stent.. J Korean Soc Radiol 2012 Jan;66(1): Hoevels J, Nilsson U. Intrahepatic vascular lesions following nonsurgical percutaneous transhepatic bile duct intubation. Abdominal Imaging 1980;5: Marshall MM, Muiesan P, Srinivasan P, et al. Hepatic artery pseudoaneurysms following liver transplantation: incidence, presenting features and management. Clinical radiology 2001;56: Samek P, Bober J, Vrzgula A, Mach P. Traumatic hemobilia caused by false aneurysm of replaced right hepatic artery: case report and review. The Journal of trauma 2001;51: Sato N, Yamaguchi K, Shimizu S, et al. Coil embolization of bleeding visceral pseudoaneurysms following pancreatectomy: the importance of early angiography. Archives of Surgery 1998;133: Otah E, Cushin BJ, Rozenblit GN, Neff R, Otah KE, Cooperman AM. Visceral artery pseudoaneurysms following pancreatoduodenectomy. Archives of Surgery 2002;137: Tessier DJ, Fowl RJ, Stone WM, et al. Iatrogenic hepatic artery pseudoaneurysms: an uncommon complication after hepatic, biliary, and pancreatic procedures. Annals of vascular surgery 2003;17: Khan AN, Fitzgerald S, Sherlock D, Tam E. Sonographic diagnosis and follow up of idiopathic hepatic artery aneurysm, an unusual cause of obstructive jaundice. Journal of Clinical Ultrasound 2001;29: Messina LM, Shanley CJ. Visceral artery aneurysms. Surgical Clinics of North America 1997;77: Tobben PJ, Zajko AB, Sumkin JH, et al. Pseudoaneurysms complicating organ transplantation: roles of CT, duplex sonography, and angiography. Radiology 1988;169: Green M, Duell R, Johnson C, Jamieson N. Haemobilia. British journal of surgery 2001;88: Nicholson T, Travis S, Ettles D, et al. Hepatic artery angiography and embolization for hemobilia following laparoscopic cholecystectomy. Cardiovascular and interventional radiology 1999;22:20-24.

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