untitled

경피경간담도내시경술이후에발병한담즙흉 1 예 울산대학교의과대학 1 울산대학교병원내과학교실, 2 서울아산병원핵의학교실박찬성 1, 이순정 1, 도기원 1, 오쌍용 1, 조현 1, 김민수 1, 홍일기 2, 방성조 1, 제갈양진 1, 안종준 1, 서광원 1 A Case of Cholethorax following Percutaneous Transhepatic Cholangioscopy Chan Sung Park, M.D. 1, Soon Jung Lee, M.D. 1, Gi Won Do, M.D. 1, Ssang Yong Oh, M.D. 1, Hyun Cho, M.D. 1, Min Su Kim, M.D. 1, Il Ki Hong, M.D. 2, Sung-Jo Bang, M.D. 1, Yang Jin Jegal, M.D. 1, Jong-Joon Ahn, M.D. 1, Kwang Won Seo, M.D. 1 1 Department of Internal Medicine, Ulsan University Hospital, Ulsan, 2 Department of Nuclear Medicine, Asan Medical Center, University of Ulsan College of Medicine, Seoul, Korea Cholethorax (bilious pleural effusion) is an extravasation of bile into the thoracic cavity via a pleurobiliary fistula (and also a bronchobiliary fistula). It is an extremely rare complication of thoraco-abdominal injuries. It can be caused by congenital anomaly and also by hepatobiliary trauma, severe infection or iatrogenic procedures. The definitive diagnosis is made with aspiration of bilious fluid from the pleural space during thoracentesis, by finding a fistulous tract during endoscopic retrograde cholangiopancreatography (ERCP) or cholagioscopy, or with finding an abnormal pleural accumulation of radioisotope during hepatobiliary nuclear imaging. Its symptoms include coughing, fever, dyspnea and pleuritc chest pain. Herein we report on a case of cholethorax following performance of percutaneous transhepatic cholangioscopy (PTCS) to remove incidentally discovered common bile duct (CBD) stones. (Tuberc Respir Dis 2008;65:131-136) Key Words: Biliary fistula, Pleural effusion, Bile, Cholangiography, Technetium Tc 99m Diethyl-iminodiacetic Acid 서 담즙흉은담도또는담낭과흉강사이에형성된누공을통해담즙이흉강으로누출되어흉수의형태로관찰되는흉막염의일종을지칭하며주로흉강-복부외상과관련된매우드문합병증이다 1. 간담도부위의외상, 감염, 의인성손상등이주요원인이며천자된흉수에서담즙을확인하거나담즙객담 (biliptysis) 을확인하여진단한다. 내시경역행췌담관조영술 (endoscopic retrograde cholangiopancreatography, ERCP), 또는경피경간담도내시경술 (percutaneous transhepatic cholangioscopy, PTCS) 을이용하거나간담도스캔등의영상적방법으로흉강내로의담즙누출 Address for correspondence: Kwang Won Seo, M.D. Department of Internal Medicine, Ulsan University Hospital, University of Ulsan College of Medicine, 290-3, Jeonhadong, Dong-gu, Ulsan 682-714, Korea Phone: 82-52-250-7029, Fax: 82-52-251-8235 E-mail: kwseo@uuh.ulsan.kr Received: Jun. 18, 2008 Accepted: Jul. 14, 2008 론 이나누공을확인하여확진할수있다. 주요증상은발열, 흉통, 호흡곤란등이며기관지담도누공 (bronchobiliary fistula) 이동반된경우는담즙객담을보일수있다. 그동안국내에는담즙흉에대한문헌보고가없었으며, 저자들은최근외상후흉막성흉통과호흡곤란을호소하였고총담관결석제거를위한경피경간담도내시경술이후에다량의흉수가발생한환자에서담즙흉으로진단된환자 1예를경험하였기에문헌고찰과함께보고한다. 증 환자 : 64세, 여자주소 : 내원당일외상후발생한우측흉통및호흡곤란과거력및가족력 : 35년전담낭절제술흡연력및음주력 : 흡연력과음주력없음. 현병력 : 내원 3일전길을걸어가던중후진하던차와무릎뒷부분이부딪히면서넘어졌으며, 동시에함께넘어진사람에의해오른쪽가슴부위를눌리게된뒤우측흉통및호흡곤란발생되어외부병원입원치료받았으며 례 131

CS Park et al: A case of cholethorax following PTCS 이후상기증상지속되어본원응급실로전원되었다. 진찰소견 : 본원응급실내원당시혈압은 109/66 mmhg, 맥박 66회 / 분, 호흡수 22회 / 분, 체온 36.6 o C였다. 의식은명료하였으나급성병색을보였고결막관찰에서빈혈의증후는보이지않았다. 흡기호흡시에우측흉통을호소하였으며우측흉부에압통을호소하였으나뼈마찰음 (bony crepitus) 은없었으며, 피하출혈, 멍 (bruise) 또는부종의증거는관찰되지않았다. 흉부청진상수포음, 천명및흉막마찰음은들리지않았으며심음은정상이었다. 복부는편평하였으며압통이나반발통은없었다. 양측하지에경한압통을호소하였으나멍또는부종의증거는관찰되지않았으며정강뼈앞함요부종 (pretibial pitting edema) 은관찰되지않았다. 초기검사실소견 : 내원당시말초혈액검사에서백혈구 4,990/mm 3 ( 호중구 58.6%), 혈색소 12.6 g/dl, 헤마토크리트 37.9%, 혈소판 209,000/mm 3 이었고혈청생화학검사에서총단백질 6.6 g/dl, 알부민 4.1 g/dl, AST/ALT 57/29 IU/L, 총빌리루빈 0.9 mg/dl, 직접빌리루빈 0.3 mg/dl, BUN 13.8 mg/dl, Creatinine 0.9 mg/dl, LDH 422 IU/L ( 참고치 : 218 472 IU/L), D-dimer 0.11 μg/ml ( 참고치 : 0 1 μg/ml), Troponin T <0.010 ng/ml ( 참고치 : 0 0.10 ng/ml) 이었다. 혈청전해질검사에서 Na 144 meq/l, K 4.0 meq/l, Cl 108 meq/l 로정상소견을보였다. 혈액응고검사에서프로트롬빈시간은 12.9초 (INR 1.01), 활성부분프로트롬빈시간은 29.5초로정상범위내였다. 실내공기에서측정된동맥혈가스분석에서 ph 7.409, PaCO 2 39.9 mmhg, PaO 2 56.6 mmhg, HCO 3 24.7 mm/l, O 2 saturation 90.8% 로저산소증을보였다. 초기방사선검사소견 : 내원당일검사한단순 X-선검사에서늑골골절은관찰되지않았다. 흉부및복부전산화단층촬영상폐영상스캔에서양쪽하엽구역기관지 (subsegmental bronchi) 에수동적무기폐에의한경계가불분명한경결 (consolidation) 이관찰되었으나흉수는보이지않았다. 전조영제영상에서총담관에고감쇄물질 (high attenuation material) 로차있는소견보여총담관결석의가능성이높은소견이었으며, 담도확장및과거담낭절제소견이관찰되었다. 임상경과및추가검사 : 환자는흉통의조절및총담관결석의제거를위해입원하였으며, 환자가구강의이상감각및치통을호소하여내시경역행췌담관조영술을시행하지못하였고, 입원후 2병일에경피경간담도배액술 (percutaneous transhepatic biliary drainage, PTBD) 을 Figure 1. Follow-up tubography after tract dilatation shows total obstruction of the distal common bile duct (CBD) by a filling defect (arrow), which may be a CBD stone. 시행하였다. 5병일에경피경간담도내시경술을통한총담관결석제거를위해담도확장술및 18 프렌치의 PTCS 튜브를삽입하였고튜브끝은총담관내에위치시켰다. 이어서담관조영술을시행한결과하부총담관내의감입된총담관결석에의해조영제가십이지장으로내려가지않는소견이관찰되었다 (Figure 1). 환자는 8병일에담관조영술을시행받았으며 5병일때에시행했던담관조영술때와는달리조영제가십이지장으로내려가는소견보였으나총담관내에조영제의결손부위가관찰되었다. 환자는내원 14병일때에경피경간담도내시경술을시행받았으며, 총수담관내에더이상남아있는결석의증거는없는것으로확인되어잔존결석이빠져나간것으로판단하여시술후 PTCS 튜브를제거하였다. 환자는 PTCS 튜브를제거한다음날부터더욱심해진우측흉통및호흡곤란을호소하였으며단순흉부X-선검사에서우측흉수가발생됨을확인하였다. 이후발열및흉수증가없이흉통이다소감소되어추적관찰중내원 18병일때추적흉부 X-선검사에서흉수의양이늘고 38.5 o C의발열이있어흉부전산화단층촬영 (Figure 2) 및흉수천자를시행하였다. 흉수검사결과는 ph 7.593, 총단백질 2.4 g/dl ( 혈청총단백질 5.5 g/dl), 알부민 1.4 g/dl ( 혈청알부민 3.3g/dl), 포도당 139 mg/dl ( 혈청포도당 130 mg/dl), LDH 2192 IU/L, 백혈구 8,000/mm 3 ( 호중구 96%, 림프구 2%, 단핵구 2%), 적혈구 1,600/mm 3 로삼출액 (exudative effusion) 132

Tuberculosis and Respiratory Diseases Vol. 65. No. 2, Aug. 2008 Figure 2. Chest CT scan shows remnant transhepatic biliary fistula (black arrows) after percutaneous transhepatic cholangioscopy (PTCS) tube removal (A) and right pleural effusion (white arrows) (B). mg/dl, 아밀라아제 70 mg/dl 로담즙흉에합당한소견임을확인하였다. 내원 22병일때에 99m Tc-mebrofenin 간담도스캔을시행하여간에서생성된담즙이우측흉강내로누출되어흉수를형성하는것을확인하였다 (Figure 4). 접촉사고당시흉부의압박외상으로횡경막손상의가능성을의심하여흉강경검사를고려하였으나 pigtail 카테터삽입후환자의발열은감소하고심한통증이점차감소되는등증상이호전되었으며, 또한환자가더이상의침습적검사를원치않아검사를시행하지않았다. 환자는이후점차호소하던흉통이소실되었고담즙배액이줄었으며감염의증거를보이지않아내원 40병일에 pigtail 카테터를제거한후퇴원하게되었다. Figure 3. A bottle of fluid shows yellow-brownish bilious fluid from thoracic cavity via chest pigtail catheter. 에합당한소견으로, 부폐렴성흉수또는횡경막하농양과같은복부감염에의한이차적인흉막삼출액을의심하여광범위항생제를투여하였으며, 내원 21병일때에 pigtail 카테터삽입을시행하여지속배액을유도하였고그날총 1400 ml의흉수가배액되었다. 그러나환자의흉강내에서배액되는흉수의색깔이담즙과비슷한황갈색소견을보여 (Figure 3), 담즙흉을의심하였고흉수생화학검사를시행하였다. 흉수검사에서총빌리루빈 21.4 mg/dl ( 혈청총빌리루빈 1.2 mg/dl), 직접 / 간접빌리루빈 11.7/9.7 고 담즙흉은 1850년도 Peacock에의해간포충증 (hepatic echinococcosis) 을진단받은 20세여성에게서병발한기관지담도누공의예로처음문헌보고되었다 (Dasmahapatra 등 2 에의해인용됨 ). 이후여러발생원인이보고되었으며담도흉강누공에는기관지담도누공을포함해서보고되고있는데, 선천적기형 3 의보고가있으며후천적원인으로는외상과관련되어간외상 1, 복부둔기외상 4, 흉-복부의총기손상 (gunshot injury) 5,6 등의예가보고되었으며, 감염과관련되어간의기생충감염 7, 간아메바농양 8, 담낭질환과관련된담낭흉강누공 (cholecystopleural fistula) 9 등이있으며의인성원인으로는담도스텐트의위치이동 찰 133

CS Park et al: A case of cholethorax following PTCS Figure 4. Hepatobiliary scan using 370 MBq of 99m Tc-mebrofenin shows the radioactivity in the right pleural space after 30 minutes of injection in anterior view (A) and right lateral view (B) (arrows). After opening the clamp of pigtail catheter which was inserted into the right pleural space, drainage of the accumulated radioactivity is noted (arrowhead). (biliary stent migration) 2, 고주파절제 (radio-frequency ablation) 10, 담낭절제술 11, 간조직검사 12, 경도관동맥색전술 (transcatheter arterial embolization) 13, 경피경간담도배액술 14, 경피경간담낭배액술 (percutaneous transhepatic gallbladder drainage, PTGBD) 15, 경피경간담도내시경술 16 그리고담도수술후담도협착과동반 17 등과같이시술또는수술이후에합병증으로병발한예들이보고되었다. 그외횡경막손상없이직접적으로횡경막을통해담즙이통과되어담즙흉이발생된예도있었다 18. 이는복수가흉강내로이동하여발생하는간성물가슴증 (hepatic hydrothorax) 의발생과유사한기전으로설명되었다. 담즙흉의증상은기침 10, 흉부압박감 19, 호흡곤란 15,16,18,20, 흉막성흉통 9,15,20, 발열 6,10,15,16, 어깨나견갑골부위로의방사통 16 등이보고되었으며염증성흉수의일반적인증상인호흡곤란과발열및흉통이흔하다. 그외누공이기도나기관지와동반되거나단독으로연결되어있는기관지담도누공의경우에는담즙객담이관찰될수있으며이경우진단에특징적인증거 (pathognomonic hallmark) 이다 6. 담즙은화학자극을일으킬수있으며흉막염증및흉막유착을일으킬수있어가능한빠른시간에흉강배액등을통해제거해주는것이추천된다 20. 진단은흉수천자에서담즙의존재를통해확진되는데생화학검사를통해 흉수-혈청총빌리루빈의비 (pleural- serum total bilirubin ratio) 가 1을초과할때진단할수있다 5. 또한담도흉강누공또는담도기관지누공을확인하기위해내시경역행췌담관조영술또는경피경간담도내시경술을이용하거나간담도스캔을통한담즙의흉강내누출을확인하는방법이있다 5,19. 간담도스캔은대개 99m Tc-iminodiacetic acid를이용하여시행하며비교적저렴한비용과쉬운방법의진단방법으로알려져있다. 그러나손상의범위를파악하거나누공의위치파악에는한계가있다 5. 담즙흉의치료로는본증례에서와같이흉관또는 pigtail 카테터를이용한흉강배액을하면서담즙흉이호전되는경우로이와같은보존적치료로완치가되기도하며 15, 담도폐쇄가동반되었을경우에는치료적인조임근절개술 (sphincterotomy), 스텐트삽입을시도하거나 6 다른경로를통해 16 감압을해주는것이추천된다. 그러나담관손상이심하거나보존적인치료로실패한경우, 조절되지않는폐나흉강내의염증, 호흡부전의진행등이동반된경우에서는수술적치료를고려할것을추천하고있다 6. 본증례와유사하게 De Meester 등 16 은경피경간담도내시경술이후에발생된담즙흉을보고하였는데담관염 (cholangitis) 이합병된담도결석 (choledocholithiasis) 환 134

Tuberculosis and Respiratory Diseases Vol. 65. No. 2, Aug. 2008 자에서경피경간담도내시경술을시행하여결석제거를성공적으로치료한뒤퇴원했던환자가 3개월뒤에발생된황달, 발열, 호흡곤란, 우측견갑골부위통증및흉수로내원하였고재입원후시행한내시경역행췌담관조영술에서담관결석및담도흉강누공을확인하여확진하였던예였다. 그환자는비담도관 (nasobiliary catheter) 을우측간담도 (hepatic duct) 에삽입한뒤반복내시경역행췌담관조영술에서담관결석및누공의소실을확인하게되었으며천자된흉수에서담즙확인및 E. coli 균이배양되어항생제및흉강배액관으로배액치료받았다. 저자는다른경로의효과적인담즙배액이치료성공에중요하다고제시하였다. 본증례에서의담즙흉의발생에대한추론으로일련의교통사고와연관된외상에의한횡경막의손상이있었을가능성과횡경막파열등의손상은없는상태에서경피경간담도내시경술과관련된누공에서누출된담즙이환자가원래가지고있던횡경막결손을통하여흉강으로이동한경우를생각해볼수있는데, 저자들은전자의가능성이더높을것으로추정하였다. 그이유로는후진하는차에접촉되어환자가넘어진후또다른사고피해자에의해우측가슴부위를눌리면서당시심한흉통을호소하게되었고늑골골절이나흉수의발생은없었지만이후에도증상호전이적었던점, 경피경간담도배액술및담도내시경시술과정에있어서횡경막을통과하여담도로누공을형성하거나하는의인성의횡경막손상이관찰되지않았던점 (Figure 1, 2) 등을들수있다. 즉, 외상에의해환자의우측횡경막이전산화단층촬영영상에서확인되지않을정도의국소파열이동반되어있다가 PTCS 튜브를제거했을때인공적누공으로흘러나온담즙이파열된횡경막을통해누출되었을가능성이다. 당시저자들은정확한원인규명을위해흉강경검사시행을고려하였으나 pigtail 카테터유치후담즙흉의호전과환자의원치않음으로인해시행하지않아정확한감별적분석은하지못하였다. 또한경피경간담도내시경술을위해서는진행적인담도확장 (progressive duct dilatation) 과약 2주간누공의성숙 (maturation) 이필요하다 16. 따라서장시간튜브유치로인해누공의자연적막힘이더디게되면서본환자의담즙흉의발생과유지에영향을미쳤을것으로사료된다. 담즙흉은주로흉강-복부외상과관련된매우드문합병증이며특히의인성원인인간담도질환의시술및수술과관련되어발생할수있어간담도계의시술및수술이더욱보편화되어시행되고있는최근에그발생이늘어날가능 성이있을것으로사료된다. 그러나그동안국내에는담즙흉에대한문헌보고가없었으며, 저자들은최근외상후흉막성흉통과호흡곤란을호소하였고담도결석제거를위한경피경간담도내시경술이후에흉수가발생한한환자에서담즙흉을진단하고성공적으로치료한경험이있어이를보고하는바이다. 요 담즙흉은담도또는담낭과흉강사이에형성된누공을통해담즙이흉강으로누출되어흉수의형태로관찰되는흉막염의일종으로, 주로흉강-복부외상과관련된매우드문합병증이며그동안국내에는담즙흉에대한문헌보고가없었다. 이에저자들은최근외상후흉막성흉통과호흡곤란을호소하였고담도결석제거를위한경피경간담도내시경술이후에흉수가발생한환자에서담즙흉으로진단된환자 1예를경험하였기에보고한다. 약 참고문헌 1. Franklin DC, Mathai J. Biliary pleural fistula: a complication of hepatic trauma. J Trauma 1980;20:256-8. 2. Dasmahapatra HK, Pepper JR. Bronchopleurobiliary fistula. A complication of intrahepatic biliary stent migration. Chest 1988;94:874-5. 3. Weitzman JJ, Cohen SR, Woods LO Jr, Chadwick DL. Congenital bronchobiliary fistula. J Pediatr 1968;73: 329-34. 4. Brunaud L, Sebbag H, Bresler L, Tortuyaux JM, Boissel P. Left hepatic duct injury and thoracobiliary fistula after abdominal blunt trauma. Hepatogastroenterology 2000;47:1227-9. 5. Feld R, Wechsler RJ, Bonn J. Biliary-pleural fistulas without biliary obstruction: percutaneous catheter management. AJR Am J Roentgenol 1997;169:381-3. 6. Navsaria PH, Adams S, Nicol AJ. Traumatic thoracobiliary fistulae: a case report with a review of the current management options. Injury 2002;33:639-43. 7. Amir-Jahed AK, Sadrieh M, Farpour A, Azar H, Namdaran F. Thoracobilia: a surgical complication of hepatic echinococcosis and amebiasis. Ann Thorac Surg 1972;14:198-205. 8. Roy DC, Ravindran P, Padmanabhan R. Bronchobiliary fistula secondary to amebic liver abscess. Chest 1972; 62:523-4. 135

CS Park et al: A case of cholethorax following PTCS 9. Delco F, Domenighetti G, Kauzlaric D, Donati D, Mombelli G. Spontaneous biliothorax (thoracobilia) following cholecystopleural fistula presenting as an acute respiratory insufficiency. Successful removal of gallstones from the pleural space. Chest 1994;106:961-3. 10. Pende V, Marchese M, Mutignani M, Polinari U, Allegri C, Greco R, et al. Endoscopic management of biliopleural fistula and biloma after percutaneous radiofrequency ablation of liver metastasis. Gastrointest Endosc 2007;66:616-8. 11. Lehur PA, Guiberteau-Canfrère V, Bury A, Cloarec D, Le Borgne J. "Cholethorax" revealing injury to the common bile duct after celioscopic cholecystectomy. Ann Chir 1992;46:450-2. 12. Pisani RJ, Zeller FA. Bilious pleural effusion following liver biopsy. Chest 1990;98:1535-7. 13. Ichikawa T, Yamada T, Takagi H, Abe T, Ito H, Sakurai S, et al. Transcatheter arterial embolization-induced bilious pleuritis in a patient with hepatocellular carcinoma. J Gastroenterol 1997;32:405-9. 14. Armstrong CP, Taylor TV. Intrapleural leakage of bile complicating percutaneous transhepatic drainage of the obstructed biliary tree. J R Coll Surg Edinb 1982; 27:308-9. 15. Lee MT, Hsi SC, Hu P, Liu KY. Biliopleural fistula: a rare complication of percutaneous transhepatic gallbladder drainage. World J Gastroenterol 2007;13:3268-70. 16. De Meester X, Vanbeckevoort D, Aerts R, Van Steenbergen W. Biliopleural fistula as a late complication of percutaneous transhepatic cholangioscopy. Endoscopy 2005;37:183. 17. Boyd DP. Bronchobiliary and bronchopleural fistulas. Ann Thorac Surg 1977;24:481-7. 18. Rowe PH. Bilothorax: an unusual problem. J R Soc Med 1989;82:687-8. 19. Lee JK. Tc-99m DISIDA hepatobiliary scintigraphy showing bile leakage into the thoracic cavity. Clin Nucl Med 2001;26:861-2. 20. Turkington RC, Leggett JJ, Hurwitz J, Eatock MM. Cholethorax following percutaneous transhepatic biliary drainage. Ulster Med J 2007;76:112-3. 136