DOI: 10.4046/trd.2011.70.3.261 ISSN: 1738-3536(Print)/2005-6184(Online) Tuberc Respir Dis 2011;70:261-265 CopyrightC2011. The Korean Academy of Tuberculosis and Respiratory Diseases. All rights reserved. 원인불명의담즙흉 1 예 Case Report 충북대학교의과대학 1 내과학교실, 2 흉부외과학교실성문혁 1, 김성무 1, 유숙희 1, 박우리 1, 안진영 1, 최강현 1, 이기만 1, 김시욱 2 A Case of Cholethorax Developed by Unknown Cause Mun Hyuk Seong, M.D. 1, Sung Moo Kim, M.D. 1, Suk Hee Yoo, M.D. 1, Woo Ri Park, M.D. 1, Jin Young An, M.D. 1, Kang Hyeon Choe, M.D. 1, Ki Man Lee, M.D. 1, Si Wook Kim, M.D. 2 Departments of 1 Internal Medicine, 2 Thoracic and Cardiovascular Surgery, Chungbuk National University College of Medicine, Cheongju, Korea Cholethorax is a bilious pleural effusion caused by a pleurobiliary fistula or leakage of bile into the pleural space. Most cases of cholethorax arise from a complication of abdominal trauma, hepatobiliary infection, or invasive procedures or surgery of hepatobiliary system. However, we experienced a case of a patient with cholethorax of unknown origin. There was no evidence of pleurobiliary fistula or leakage of bile from the hepatobiliary system although we examined the patient with various diagnostic tools including chest and abdominal computed tomography, endoscopic retrograde cholangiopancreatography, tubography, bronchofiberscopy, hepatobiliary scintigraphy and video-assisted thoracoscopic surgery. Herein we report a case of cholethorax for which the specific cause was not identified. The patient was improved by percutaneous drainage of pleural bile. Key Words: Bile; Biliary Fistula; Pleural Effusion; Diaphragm 서 담즙흉 (cholethorax, biliothorax) 은담도계와흉막강과의비정상적인연결로인한흉강담도루 (pleurobiliary fistula) 에의해담즙이흉막강내에고이는드문질환으로주로간이나담도계의감염, 복부외상, 간이나담도계수술혹은침습적시술등의합병증으로발생하는질환이다 1-4. 또한담도계와기관지가연결되어기관지담도루 (bronchobiliary fistula) 가형성되면진단적특징인객담즙 (biliptysis) 이나오게되며 5, 담즙흉은혈청총빌리루빈치보다흉수의총빌리루빈치가높을때진단할수있다 6. 기관지담관루의국내보고는약 10예이상있으나 7 담즙흉 Address for correspondence: Ki Man Lee, M.D. Division of Pulmonology, Department of Internal Medicine, Chungbuk National University Hospital, 410, Sungbong-ro, Heungduk-gu, Cheongju 361-711, Korea Phone: 82-43-269-6353, Fax: 82-43-269-3252 E-mail: kimlee@chungbuk.ac.kr Received: Oct. 22, 2010 Accepted: Dec. 17, 2010 론 은 1예의국내보고가있었다 8. 기관지담도루나담즙흉은대부분은선행원인이있지만, 저자들은원인이불명확하고또한누공을찾기위한여러가지검사에도불구하고누공이나담즙누출을찾을수가없었던원인불명의담즙흉 1예를경험하였기에문헌고찰과함께보고하는바이다. 증례환자 : 72세, 남자주소 : 내원 6시간전부터발생한우상복부통증및호흡곤란과거력및가족력 : 10년전당뇨병진단받았으나투약없이지냈다. 사회력 : 수년전부터하루에막걸리 1병정도를마셨으며, 20갑년의흡연력이있었다. 현병력 : 내원약 6시간전부터우하부흉통및호흡곤란이발생하여본원응급실로왔다. 통증은서서히시작되었으며지속적으로있었고, 흡기시와상체를움직일때 261
MH Seong et al: A case of cholethorax Figure 1. (A) Chest X-ray shows right pleural effusion. (B) Chest CT scan shows large amount of pleural effusion with passive atelectasis. (C) Abdomen CT scan shows mild intrahepatic duct dilatation (arrows). CT: computer tomography. 악화되었다. 환자는이전에비슷한종류의통증이나호흡곤란은없었다고하였으며, 약 1개월전화장실에다녀오다미끄러져앞으로넘어진것이외의외상은없었고당시에는복부나흉부의통증을기억하지못하였다. 진찰소견 : 응급실내원당시혈압은 160/90 mm Hg, 맥박 74회 / 분, 호흡수 22회 / 분이었고체온은정상이었다. 의식은명료하였고급성병색을보였다. 이학검사에서빈혈이나황달의소견은보이지않았다. 우측호흡음이감소되어있었고, 복부는전체적으로편평하고부드러웠으며촉진시압통이나반발통은없었다. 검사실소견 : 내원당시말초혈액검사에서백혈구 8,200/mm 3 ( 호중구 77.2%), 혈색소 11.9 g/dl, 혈소판 209,000/mm 3 이었고 AST 14 IU/L, ALT 9 IU/L, γ-gt 19 IU/L, ALP 185 IU/L, 총빌리루빈 0.33 mg/dl, 아밀라제 33 IU/L, 리파제 49 IU/L, 총단백질 6,700 mg/dl, 알부민 3.9 g/dl, BUN 15 mg/dl, creatinine 1.04 mg/dl, LDH 274 IU/L. C-반응단백 (CRP) 0.46 mg/dl 였다. 실내공기에서측정한동맥산소포화도는 96% 로정상이었다. 방사선검사소견 : 내원시검사한흉부 X-선검사에서우측흉수소견 (Figure 1A) 을보였고흉부전산화단층촬영검사에서다량의우측흉수와수동적무기폐가관찰되었지만골절이나외상의흔적은없었다 (Figure 1B). 복부 Figure 2. Percutaneously needle aspirated pleural effusion was dark greenish colored fluid. 전산화단층촬영에서도경도의간내담관확장소견을보였으나그외복강내병변은없었다 (Figure 1C). 임상경과및추가검사 : 흉수검사는 ph 7.5, 단백질 9,470 mg/dl, 포도당 1 mg/dl, LDH 5,142 mg/dl의삼출액소견으로아밀라제 69 IU/L, 아데노신탈아미노효소 (adenosine deaminase, ADA) 는 125 IU/L였으며, 흉수세포검사는호중구 45%, 림프구 13%, 조직구 42% 였다. 흉수색깔이짙은녹색을띠는혼탁한액체 (Figure 2) 로담즙흉을의심하여측정한흉수의총빌리루빈은 11.7 262
Tuberculosis and Respiratory Diseases Vol. 70. No. 3, Mar. 2011 Figure 3. (A) The retrograde cholangiopanrceatography shows no evidence of stricture, obstruction of biliary tree or leakage of contrast media. (B) Retrograde cholangiography shows no evidence of biliary tree abnormality or leakage of contrast media. (C) Contrast media injected into pleural cavity via Pigtail cathether. There is no evidence of fistula or leakage of contrast media into extrapleural space. mg/dl로담즙흉에합당하였다. 추가적으로흉수에서의담즙산은 1.4 umol/l 였다. 흉수의세균배양검사에서는 3일후 Staphylococcus hominis가배양되었다. 흉수를제거하기위해 12 French Pigtail 카테터를경피적으로흉막강내에삽관하였고첫 24시간동안약 1,100 ml 정도의짙은녹색담즙이배액되었다. 입원 1일후실시한내시경역행췌담관조영술 (endoscopic retrograde cholagiopanceatography, ERCP) 에서담즙누출이나누공의소견은없었고정상이었으나일단내시경적유두괄약근절개술을실시하고담도배액관를유지하였다 (Figure 3A). 입원 3일째담도배액관을통한담관조영술을시행하였으나담도내의이상소견이나조영제의누출소견도없었다 (Figure 3B). 입원후흉막강카테터를통해서하루에 200 ml 내지 400 ml 정도의흉수가계속배출되면서흉통과호흡곤란은호전되었다. 객담즙은없었으나기관지담도루등기관지내병변을확인하기위해입원 7일째시행한기관지내시경에서도우하부기관지가흉수압력에의한것으로보이는경도의압박소견이외에기관지내병변은없었다. 입원 9일째흉막강내카테터를통해서조영제를주입하였으나조영제가담도계나복강내로누출된소견은보이지않았다 (Figure 3C). 입원 12일째카테터를통해서하루 100 ml 정도만배액이되었고환자의상태가현저히호전되었다. 횡격막 Figure 4. Video assisted thoracic surgery shows severe thickening of pleura with multiple adhesions. Star indicates right diaphragm and bile stained visceral pleura was seen at right upper lobe (triangle) and right lower lobe (arrow). 천공이나흉강내이상소견을확인하기위해시행한비디오보조흉강경수술 (Video-assisted thoracic surgery, VATS) 의소견은벽측및장측흉막비후및유착소견이보였고담즙으로착색된흉막이관찰되었다 (Figure 4). 흉막의조직병리소견은비특이적인만성염증과섬유화소견외의특이소견은없었다. 입원 25일째시행한 99m Tc-diisopropyl iminodiacetic acid (DISIDA) 간담관섬광조영술 (hepatobiliary scintigraphy) 검사도정상소견이었다. 담 263
MH Seong et al: A case of cholethorax 즙흉의원인이밝혀지지는않았지만환자의상태는호전되었고배액되는흉수의양은점차감소하여입원 28일째 Pigtail 카테터를제거하였다. 카테터를제거한후에도소량의흉수는남아있었고폐의팽창은완전히이루어지지않았으나환자는별다른증상없이입원 32일째퇴원하였다. 퇴원 1개월후외래방문시흉통이나호흡곤란등의이상증상호소는없었으나추적관찰한흉부 X-선검사에서퇴원당시보다우측흉수가다소증가되어다시흉수검사를시행하였다. 흉수는담홍색으로흉수의검사결과총빌리루빈은 1.80 mg/dl ( 혈청총빌리루빈 0.37 mg/dl) 이었고, 총단백질 4,522 mg/dl ( 혈청총단백질 6.9 g/dl), 포도당 75 mg/dl, LDH 449 IU/dL ( 혈청 LDH 310 IU/L) 이었으며, ADA는 28.4 IU/L 였고림프구가 95% 를차지하는삼출액소견이었으며결핵균및세균은배양되지않았다. 환자의증상은없었고담즙흉의호전소견은보이나흉막유착에의한폐의팽창이불충분하여추후흉막박피술을고려하기로하고현재추적관찰중이다. 고찰담즙흉은담도계와흉막강사이의누공이나담도계로부터의담즙누출에의해담즙이흉막강에고이는질환으로흉수검사로흉수의총빌리루빈이혈청보다높은경우진단할수있다 6. 선행원인으로는간담도계의감염성질환이나 1 복부외상이흔하나 9-11, 최근에는간담도계의수술이나침습적시술의합병증에의한보고가증가하고있다 3,4,12,13. 대부분의보고에서는담즙흉을야기하는선행원인이있으나본증례와같이원인이불명확한 1예가보고된바있다 8. 담즙흉의원인질환과담즙의누출이나누공을확인하기위한검사로전산화단층촬영, 내시경역행췌담관조영술, 경피경간담도조영술 (percutaneous transhepatic cholangiography) 이나방사선핵종을이용한간담관섬광조영술등이사용된다. 그외진단적혹은치료적목적으로개복술이나개흉술을실시하기도한다. 전산화단층촬영으로는간이나횡격막하농양등의감염성질환, 외상에의한간열상이나횡격막파열등을진단하는데유용하다 7-9. 누공이나담즙누출을확인하기위하여내시경역행췌담관조영술이가장보편적인검사로사용되지만 9-11, 그외경피경간담도조영술이나간담관섬광조영술검사를이용하여누공이나담즙누출을확인하기도한다 8-10. 그러나이러한검사로도담즙누출이나누공을발견하지못하 는경우나 2,11 혹은원인질환의치료를위해개복술이나개흉술을실시하여농양의제거, 누공이나횡격막파열에대한수술을하기도한다 2,5,11. 본증례에서는상기언급한여러가지검사에서도담즙흉의원인을알수가없었고누공이나담즙누출의소견을발견할수가없었다. 또한횡격막파열이나누공을확인하기위해비디오보조흉강경수술을실시하여도횡격막파열이나누공을확인할수가없었다. 환자는복부증상이없었고전산화단층촬영에서간내담관의경도확장소견외에는이상소견이없으므로개복술은실시하지않았다. 결론적으로본증례에서답즙흉의원인은환자가기억하는 1개월전심하게앞으로넘어지면서발생한복부외상에의한담즙흉이발생한것으로유추된다. 복부외상이경미한경우담즙흉은있으나간기능이정상소견을보일수가있고 5, 담즙흉의유일한국내보고에서도본증례와유사하게자동차사고이후외상에의한담즙흉의가능성을제시하였다 8. 또한간생검이후발생한담즙흉의보고에서내시경역행췌담관조영술도정상이고부검에서도누공을발견하지못한경우도있었다 13. 이러한경우담즙흉이발생하는기전은외상에의해일시적으로생긴담관내출혈에의해피떡 (blood clots) 이형성되고이로인해담관내의압력이증가하면서횡격막의미세손상을통해담즙이흉강내로누출되거나 5,11, 횡격막의느슨한결체조직이늘어나면서이들미세구조를통하여음압상태의흉막강내로답즙이누출될수가있다고하였다 14. 본증례는담도확장소견이담관내출혈에의한일시적압력상승으로발생하였을가능성이있다고생각된다. 복부외상후담즙흉이발생하는기간은평균 14일이며 38일후에담즙흉이발생된경우도있어담즙이흉강내로누출되는데는다소기일이소요될수있다 11. 그러나본증례에서와마찬가지로답즙흉이일단발생하면빠르게흉막비후가진행한다고보고되었다 2. 본증례의흉수에서배양된 Staphylococcus hominis는피부에상주하는 Coagulase-Negative Staphylococci의일종으로배양이되더라도대부분의경우오염에의한것이며 15 담즙흉의원인균으로보고된증례도없어흉수검사시의오염에의한것으로생각되나이차감염의가능성도있을것으로생각된다. 담즙흉의증상은일반적인흉막염의증상과동일하나만일담도계와기관지의연결에의한기관지담도루가생기면특징적진단소견인객담즙이나타나며, 이경우담즙에의한기관지염증및괴사성폐렴등을동반하게되 264
Tuberculosis and Respiratory Diseases Vol. 70. No. 3, Mar. 2011 어담즙흉에비해더욱중한경과로사망률이높다 7. 그러나담즙흉만있는경우는원인질환이나간및췌담도계손상의정도에따라다양한예후를갖는다. 담즙흉의치료는원인질환및누공의유무와위치에따라다르며심한간손상이나간담도계나횡격막하농양이나누공이있을경우에는일반적으로개흉술이나개복술이추천된다 1,2,5,11. 그러나본증례와같이간담도계질환이나손상이비교적경미하고누공이없는경우는보존적치료만으로도호전될수도있다. 암등에의한담도협착이있는경우는흉관을통한흉수배액과함께경피담도배액혹은내시경적담도감압술로호전되었으며 3, 또한담도협착이없더라도담도감압술이담도계의압력을낮추어담즙의흉강내이동을줄여담즙흉이호전된다고보고하였다 11. 또한경미한외상의경우흉강내가슴관이나카테터삽관에의한흉수배액만으로도환자가호전됨을보고하였다 9-11. 본증례에서도원인질환을찾을수없었고여러검사를시행하였음에도불구하고담즙누출이나누공을찾을수가없었으며, 내시경적유두괄약근절개술및흉막강내카테터삽관으로흉수를배액하면서증상이호전된담즙흉 1예를경험하였기에이를보고하는바이다. 참고문헌 1. 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. 2. Franklin DC, Mathai J. Biliary pleural fistula: a complication of hepatic trauma. J Trauma 1980;20:256-8. 3. Strange C, Allen ML, Freedland PN, Cunningham J, Sahn SA. Biliopleural fistula as a complication of percutaneous biliary drainage: experimental evidence for pleural inflammation. Am Rev Respir Dis 1988;137:959-61. 4. 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. 5. Oparah SS, Mandal AK. Traumatic thoracobiliary (pleurobiliary and bronchobiliary) fistulas: clinical and review study. J Trauma 1978;18:539-44. 6. Light RW, Lee G. Textbook of pleural diseases. 2nd ed. London: Hodder Arnold; 2008. 7. Lee SH, Lee KJ, Kim SY, Lee SK, Jung KS, Park BH, et al. Bilioptysis caused by bronchobiliary fistula secondary to sclerosing therapy of liver cyst. Tuberc Respir Dis 2010;69:119-23. 8. Park CS, Lee SJ, Do GW, Oh SY, Cho H, Kim MS, et al. A case of cholethorax following percutaneous transhepatic cholangioscopy. Tuberc Respir Dis 2008;65: 131-6. 9. Feld R, Wechsler RJ, Bonn J. Biliary-pleural fistulas without biliary obstruction: percutaneous catheter management. AJR Am J Roentgenol 1997;169:381-3. 10. Sheik-Gafoor MH, Singh B, Moodley J. Traumatic thoracobiliary fistula: report of a case successfully managed conservatively, with an overview of current diagnostic and therapeutic options. J Trauma 1998;45:819-21. 11. 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. 12. Rowe PH. Bilothorax--an unusual problem. J R Soc Med 1989;82:687-8. 13. Pisani RJ, Zeller FA. Bilious pleural effusion following liver biopsy. Chest 1990;98:1535-7. 14. Córdoba López A, Monterrubio Villar J, Bueno Alvarez- Arenas I. Biliothorax unrelated to fistula: a rare complication in gallbladder disease. Arch Bronconeumol 2008;44:396-7. 15. Sewell CM, Clarridge JE, Young EJ, Guthrie RK. Clinical significance of coagulase-negative staphylococci. J Clin Microbiol 1982;16:236-9. 265