The Korean Journal of Gastrointestinal Endoscopy Case Report 악성십이지장 - 대장루 1 예 김세원ㆍ채현석ㆍ민정요ㆍ손혜숙ㆍ김진수ㆍ김형근ㆍ조영석ㆍ안창혁 * 가톨릭대학교의과대학내과학교실, * 외과학교실 A Case of Malignant Duodenocolic Fistula Sei Won Kim, M.D., Hiun Suk Chae, M.D., Jeong Yo Min, M.D., Hye Suk Son, M.D., Jin Su Kim, M.D., Hyung Keun Kim, M.D., Young Seok Cho, M.D. and Chang Hyuk Ahn, M.D.* Departments of Internal Medicine and *Surgery, College of Medicine, The Catholic University of Korea, Seoul, Korea Malignant duodenocolic fistula is a rare complication of colon cancer, and this usually develops as the right-side colon cancer or colonic hepatic flexure cancer infiltrates into the second portion of the duodenum. Six Korean cases of this malignancy have been previously reported on. The patients usually complain of watery diarrhea, feculant vomiting and weight loss that can be attributed to the altered normal flora. Barium enema has been the diagnostic procedure of choice to demonstrate the fistulous tract, but with the technical development of gastroendoscopy, the primary procedure is also changing. Curative resection is not possible in many cases. Palliative ileotransverse colostomy with gastrodjejunostomy is performed to relieve symptoms, but it cannot completely prevent the vomiting or diarrhea. We herein present a case of malignant duodenocolic fistula in a patient who had been suffering from indigestion, loose stool and feculant vomiting for one year. This case was diagnosed by endoscopy and the patient underwent a palliative operation. (Korean J Gastrointest Endosc 2009;38:38-42) Key Words: Watery diarrhea, Malignant duodenocolic fistula, Gastroduodenoscopy, Barium enema 교신저자. 채현석가톨릭대학교의과대학내과학교실 (480-130), 경기도의정부시금오동 65-1 전화 : 031-820-3028 팩스 : 031-847-2719 이메일 : minjeongyo@catholic.ac.kr 접수. 2008 년 7 월 8 일승인. 2008 년 12 월 2 일 서론 악성십이지장-결장루는대장암의매우드문합병증으로대부분횡행결장이나결장간만곡부의종양이십이지장후구부를침범하여발생한다. 1 국내에서는현재까지십이지장-장관누공증례로 42예가보고되었으며, 이중악성십이지장-대장누공증례는 6예이다. 누공을통해대장에서소장으로역류가발생함에따라장내세균총이변화하게되고, 음식물의통과시간이짧아져설사와악취가나는구토, 현저한체중감소등이주요증상으로나타난다. 2-4 전통적으로대장조영술이진단에가장유효하다고알려져있으나최근상부위장관내시경의일반화및기술발달로인해진단방법이달라진양상을보인다. 5 치료는환자상태및절제가능성에따라결정되는데광범위절제술만이생존기간연장에도움이되나실제로는대부분국소침윤이나전이가동반되어있어증상호전을위한고식적수술을시행하게되며이러한경우평균여명은 1년미만이 다. 1,6,7 저자들은 1년이상소화불량과무른변그리고악취를동반한구토를주소로내원한환자에서상부위장관내시경을시행하여십이지장후구부에상행결장으로연결되는누공을발견하였고, 병변의조직검사및병기설정을위한검사에서전이를동반한악성십이지장-결장누공으로진단하고고식적수술을시행한 1예를경험하여문헌고찰과함께보고한다. 증례 78세남자가 1주전부터심해진구역과구토, 식욕부진을주소로내원하였다. 약 1년이상소화불량과악취를동반한오심, 구토, 식욕부진이지속되면서 10 kg의체중감소와무른변을보는증상이동반되었다. 과거병력에서특이사항은없었으며, 사회력에서매일소주 1 2병을마시다 1년전부터금주중이었고 15갑년의흡연력이있었다. 가족력에서는특이사항없었다. 내원당시혈압은 120/70 mmhg, 맥박 100회 / 분, 호흡 38 The Korean Journal of Gastrointestinal Endoscopy
수 20회 / 분, 체온 37.2 o C였다. 진찰소견에서만성병색소견보였으며, 결막이창백하였고, 복부청진에서는특이소견없었다. 혈액혈구검사에서백혈구 5,100/mm 3, 혈색소 6.1 g/dl, 혈소판 421,000/mm 3 이었고, 생화학검사에서총단백 4.7 g/dl, 알부민 2.6 g/dl, 총빌리루빈 0.24 mg/dl, AST 26 U/L, ALT 19 U/L, ALP 232 IU/L, BUN 28.0 mg/dl, creatinine 0.77 mg/dl, Na 129 meq/l, K 4.3 meq/l, 공복혈당 116 mg/dl였으며, 혈액응고검사에서 PT (INR) 1.05이었다. 요검사는정상이었다. 종양표지자검사에서 AFP 1.19 ng/ml, CA 19-9 9.60 U/mL, CEA 2.51 ng/ml이었다. 흉부방사선사진에서기종성폐양상을보이고있었고, 복부방사선사진에서는이상소견이관찰되지않았다. 빈혈과전해질불균형교정후상부위장관내시경을실시하였다. 상부위장관내시경에서십이지장제 2부에누공을둘러싸고있는종괴가발견되었고, 누공을통해내시경을진입시킨후상행결장및맹장과회맹판을관찰할수있었다 (Fig. 1). 종괴에서조직검사를시행하였다. 복부단층촬영에서위전정부와대장간만곡부의변형과함께장관주변복강에만성염증성또는침윤성종양에의한것으로생각되는액체저류소견이관찰되었다 (Fig. 2). 다음날상부위장관조영술을실시하였다. 십이지장제 2부에서내강협착을동반한 4 cm 크기의점막손상이관찰되어십이지장종괴를시사하였고, 종괴로추측되는부위외측과상행결장사이에연결관이관찰되어누공이형성된것으로생각되었다. 연결관에인접한상행결장에도내강의협착및장벽의비후가관찰되어종양의침습가능성을시사하였다 (Fig. 3). 바륨대장조영술에서도상행결장에서위, 십이지장으로의조영제유입이확인되었다 (Fig. 4). 십이지장에서시행한조직검사에서는선암으로진단되었다. 병기설정을위한양전자단층촬영에서상행결장및양폐야의폐문부림프절에섭취율이증가하여전이성병변이의심되었다. 환자는폐기능검사에서 FEV1 34% 로중증폐쇄성폐기능장애가있었고전이성병변도의심되었기에, 고식적수술을시행하였다 (Fig. 5). 위-공장문합및회장-횡행결장문합술을실시하였 Figure 1. EGD findings. It reveals duodenal mass with a opening at the second portion. After passing the scope through the fistua, the ascending colon including the cecum with the appendiceal opening is shown. Vol. 38, No. 1 January, 2009 (38-42) 39
Figure 2. Abdomen CT findings. They show a deformity of gastric antrum and hepatic flexure (arrows) with mild fluid lining in the interloopal space, maybe resulted from chronic inflammation or malignant infiltration. There is no definite massforming lesion. Figure 3. Upper gastrointestinal series. It shows a duodenal mass at the second portion and a fistula formation with the ascending colon. The tumor invasion at the distal ascending colon is also suspected. 고현재약 10 개월간외래에서추적관찰중이다. 고찰 십이지장-결장루는매우드문질환으로원인이매우다양하여십이지장궤양천공, 크론병, 담석증, 십이지장게실염그리고췌장가성낭종파열등의양성질환과간만곡부결장암, 십이지장암그리고담낭암등의악성질환에의해발생한다. 1 악성병변에의한십이지장-결장루의경우암이인접장기에직접침윤되면서궤양및괴사를일으켜누공을형성한다. 6 1862 년 Haldane 8 이간만곡부결장암에의한십이지장-결장루에대해처음발표한이후 1982년 Chang 등 2 은문헌조사에서악성십이지장-결장루의빈도가전세계적으로 64예로전대장암의 Figure 4. Barium enema. There is a luminal narrowing and mucosal destruction lesion in the ascending colon at the hepatic flexure. And there is contrast filling in the duodenum and the stomach, probably due to the known fistula. 0.001% 에서발생하며대부분의경우횡행결장이나간굴곡부결장에생긴종양이십이지장의후구부를침범하여발생하는것으로발표하였다. Welch 등도대장암환자 900명당 1명의빈도로발생빈도가매우낮음을보고하였다. 9 국내에서는현재까지십이지장-장관누공증례로 42예가보고되었으며, 이중악성십이지장-대장누공증례는 6예이다 (Table 1). 5 십이지장-결장루의증상으로는주로설사와구토그리고현저한체중감소를들수있다. 2 설사의기전은결장에서소장으로내용물이역류함에따라장내세균총이변화하여감염성장염이발생하거나담즙산혹은염산이대장점막을자극하면서또는소장-결장단락을통한장내통과시간단축에의한것으로, 결과적으로영양실조에이르게된다. 3,4 또한환자들은대 40 The Korean Journal of Gastrointestinal Endoscopy
변양상의토물을보이고트림시에악취를호소하기도하며드물게위장출혈이동반되기도한다. 1,10 본증례에서는설사와악취가나는구토, 체중감소모두발생하였다. 기본적인이학적검사에서일차적으로복부의종물이촉지되고, 영양결핍에따른공막의창백, 전신쇄약의소견이관찰되며, 혈액검사에서전해질불균형, 저단백질혈증, 철분결핍성빈혈이보일수있다. 10 Figure 5. Operative finding. It shows adhesion of right colon to retroperitoneum around the hepatic flexure. Direct invasion to the duodenal 2nd portion is also found. On gross finding, there is no lesion compatible to carcinomatosis peritonei. 내시경으로누공의입구로의심되는병변이발견된경우누공을확인하는방법으로는방사선학적검사가유용하다. 대장조영술을통해바륨이대장에서십이지장으로유입되는것을관찰하거나상부위장관조영술에서도누공형성을확인할수있는데, 대장내강의높은압력이누공을통한조영제유입을보다용이하게하므로전자가후자보다진단율이높다. 9,11 상부위장관내시경검사는누공의진단보다는누공의원인이종양인지염증성병변인지감별하는데유용하다고알려져있으나, 최근에는내시경검사가일차검사로일반화되고내시경기술이점점발전함에따라진단방법에있어서도이전과는다소다른양상을보인다. 5 복부단층촬영은종양의국소적침윤이나전이성병변을확인하는데의의가있다. 1 본증례에서는복부단층촬영에서뚜렷한종괴형성이보이지않고위전정부와간만곡부결장의변형형태로만관찰되어병기설정을위해양성자방출촬영까지시행하였던경우로그결과폐문부의전이가의심되었다. 본증례에서는내시경을먼저시행하여십이지장에서대장으로연결된누공발견후대장조영술및상부위장관조영술로이를확진하였는데내시경을통해진단뿐아니라조직학적확진까지할수있었던경우이므로향후누공진단과정에서는조영술이차지할비중이점차감소할것으로생각한다. 십이지장-결장루의치료는원발암의범위, 전이병변의유무그리고환자상태에따라결정된다. 1 대부분의환자에서흡수장애로인해영양결핍을보이므로수술전경정맥영양요법으로영양상태개선및전해질교정에주의가필요하다. 12,13 Table 1. Review of Malignant Duodenocolic Fistula Cases in Korea Reference No. Age/ Sex Symptom Diagnostic study Treatment 15 57/M RUQ discomfort UGIS: filling defect in descending Resection of duodenum and right duodenum and irregular contrast leakage colon and adjuvant chemotherapy 6 56/M Persistent diarrhea, UGIS: barium collection between Extended right hemicolectomy and Palpable RUQ mass duodenum and right colon feeding gastrostomy BE: duodenocolic fistula 16 55/M Palpable RUQ mass, BE: apple-core sign at the hepatic flexure Extended right hemicolectomy, Diarrhea and duodenocolic fistula metastatic lymph node (-), EGD - duodenal ulcerofungating mass distant metastasis (-) with fistulous tract 17 76/F Abdominal BE: apple core sign at the transverse Extended right hemicolectomy discomfort colon and duodenocolic fistula 17 53/M Epigastric pain, EGD: duodenal ulcer with fistula Follow up loss Acute diarrhea BE: apple core sign at the right colon 5 78/F Chronic diarrhea, EGD: ulcer with a fistulous opening TPN, Palliative gastrojejunostomy Fecalous vomiting in the duodenal second portion and ileocolostomy, Discharge Colonoscopy: a fungating mass near with improved symptom the hepatic flexure RUQ, right upper quadrant; UGIS, upper gastrointestinal series; BE, barium enema. Vol. 38, No. 1 January, 2009 (38-42) 41
절제가능성이있는경우우측반대장절제및누공절제, 십이지장부분절제술후회장과횡행결장을연결하는광범위절제술을시행한다. 2 본증례와같이전이병변으로인해절제가능성이없는경우증상호전을위해회장-횡행결장문합술및위-공장문합술을시행하는데설사와구토를완전히소실시키지는못하는것으로알려져있다. 6 예후역시진단당시병의진행정도에따라달라지며고식적수술을시행할경우평균여명은 1년미만이다. 7 본증례의경우에는종괴의조직검사에서선암이진단되었고복부단층촬영과양성자방출단층촬영에서전이성병변이의심되어고식적수술만을시행후외래에서 10개월간추적관찰중이다. 비록매우드문질환이기는하나진단당시의병기가예후에중요하고내시경적진단의접근성및정확도가높으므로, 앞서기술한증상들을보이는환자들에서감별진단을위한조기내시경시행이도움이되겠다. 14 요약 악성십이지장-결장루는대장암의매우드문합병증으로국내에서는현재까지 6예가보고되어있다. 대부분횡행결장이나결장간만곡부의종양이십이지장후구부를침범하여발생한다. 장내세균총변화와관련하여수양성설사와악취가나는구토, 현저한체중감소등이주요증상으로나타난다. 대장조영술이진단에가장유용하다고알려져있으나최근에는상부위장관내시경의일반화및기술발달추세로인해진단방법이달라진양상을보인다. 치료는대부분의경우국소침윤이나전이가동반되어있어증상호전을위한고식적수술을시행하게되며구토와설사가완전히호전되기는어렵다. 저자들은 1년이상소화불량과무른변그리고악취를동반한구토를주소로내원한환자에서상부위장관내시경을시행하여십이지장후구부와상행결장간의누공을발견한후고식적수술을시행한증례를경험하여문헌고찰과함께보고한다. 색인단어 : 수양성설사, 악성십이지장-대장누공, 상부위장관내시경, 대장조영술 참고문헌 1. Soulsby R, Leung E, Williams N. Malignant colo-duodenal fistula; case report and review of literature. World J Surg Oncol 2006;4:86. 2. Chang AE, Rhoads JE. Malignant duodenocolic fistulas: a case report and review of the literature. J Surg Oncol 1982;21: 33-36. 3. Abcarian H, Udezue N. Coloenteric fistulas. Dis Colon Rectum 1978;21:281-286. 4. Torosian MH, Zins JE, Rombeau JL. Malignant colojejunal fistula: case report and review of malignant coloenteric fistula. Dis Colon Rectum 1982;25:222-224. 5. Park JM, Chung WC, Lee KM, et al. Case of malignant duodeno-colic fistula showing typical endoscopic findings. Korean J Gastrointestinal Endosc 2005;31:414-418. 6. Lee WB, Chung KS, Lee KC. Malignant duodenocolic fistula: report of a case. J Korean Surg Soc 1988;35:488-493. 7. Izumi Y, Ueki T, Naritomi G, Akashi Y, Miyoshi A, Fukuda T. Malignant duodenocolic fistula; report of a case and considerations for operative management. Surg Today 1993; 23:920-925. 8. Haldane DR. Case of cancer of the caecum, accompanied by with caecoduodenal and caecocolic fistulae. Edinburgh Med J 1862;7:624-629. 9. Welch JP, Warshaw AL. Malignant duodenocolic fistulas. Am J Surg 1997;133;658-661. 10. Vieta JO, Blanco R, Valentini GR. Malignant duodenocolic fistulas: report of two cases, each with one or more other synchronous gastrointestinal cancers. Dis Colon Rectum 1976; 19:542-552. 11. Martinez LO, Manheimer LH, Casal GL, Lubin J. Malignant fistulae of the gastrointestinal tract. Am J Roentgenol 1978;131:215-218. 12. Steer ML, Glotzer DJ. Colonic exclusion bypass principle: its use in the palliative treatment of malignant duodenocolic and gastrocolic fistulas. Arch Surg 1980;115:87-89. 13. Iuchtman M, Zer M, Plavnick Y, Rabinson S. Malignant duodenocolic fistula. The role of extended surgery. J Clin Gastroenterol 1993;16:22-25. 14. Roberts PL, Coller JA, Corriveau S, Nielsen-Whitcomb FF. Malignant duodenocolic fistula diagnosed by endoscopy. Surg Endosc 1989;3:112-114. 15.Park HB, Kim KS, Yang YC, Yeo HS. A case of colon carcinoma invading duodenum. Korean J Gastroenterol 1986; 18:223-226. 16. Kim WJ, Rim KS. A case of malignant duodenocolic fistula diagnosed by endoscopy. Korean J Gastrointest Endosc 1989; 9:203-205. 17. Koo JH, Lee KS, Cho YU, et al. Malignant duodenocolic fistulas: report of 2 cases. Korean Soc Coloproctol 2001;17: 53-57. 42 The Korean Journal of Gastrointestinal Endoscopy