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1 대한소화기내시경학회지 2008;37: 수술전대장내시경정복을시도한양성및악성병변에의한회결장형장중첩증 2 예 연세대학교원주의과대학내과학교실, * 병리과교실 이일영ㆍ김재우ㆍ예창진ㆍ채명훈ㆍ성중경ㆍ석기태ㆍ백순구ㆍ조미연 * Two Cases of Benign and Malignant Lesion Caused Ileocolic Intussusception: Preoperative Colonoscopic Reduction was Attempted for These Patients Il Young Lee, M.D., Jae Woo Kim, M.D., Chang Jin Yea, M.D., Myeong Hun Chae, M.D., Joong Kyung Sung, M.D., Ki Tae Suk, M.D., Soon Koo Baik, M.D. and Mee Yon Cho, M.D.* Departments of Internal Medicine and *Pathology, Yonsei University Wonju College of Medicine, Wonju, Korea 장중첩증은주로소아에서흔히발생하며뚜렷한원인이없이발생하는경우가많다. 이에반해성인의장중첩증은매우드물게발생하는질환이며기질적인원인이있는경우가대부분이다. 진단은최근전산화단층촬영이보편화된후수술전진단율이높아졌으나대부분수술과정에서이루어지고있다. 치료방법에는현재대부분의보고들에서개복술을필수적으로생각하고있으나뚜렷하게정립되어있는방법은아직없다. 최근소수의환자이지만대장내시경으로장중첩증을정복했다는보고가있고, 수술을시행하더라도회결장형장중첩증의경우중첩된회장이매우긴경우가많아정복을먼저시도해본다면과도한절제를막는데도움을줄수있다고알려져있다. 이에저자들은비록정복은되지못했으나양성및악성병변에의해장중첩증이발생한성인환자에서수술전대장내시경으로장중첩증을진단하고내시경적정복을시도해본환자를경험하였기에문헌고찰과함께보고한다. 색인단어 : 장중첩증, 개복술, 회결장형, 대장내시경, 정복 서 성인의장중첩증은매우드물게발생하는질환으로서대부분이악성또는양성종양의병변때문에발생한다. 전체장중첩증의 5% 가성인에서발생하고성인에서장폐쇄를일으키는모든질환중 1 5% 가장중첩증에해당된다. 1-4 진단은최근전산화단층촬영이보편화된후수술전진단율이높아졌으나대부분수술과정에서이루어지고있으며치료는유발병소를동반하기때문에개복술이필수적인것으로알려져있다. 2 그러나, 소장 론 접수 :2008 년 6 월 30 일, 승인 :2008 년 9 월 2 일연락처 : 김재우, 연세대학교원주의과대학내과학교실 ( ) 강원도원주시일산동 162 전화 : , 팩스 : 이메일 : jawkim96@yonsei.ac.kr 및우측결장에발생한경우양성유발병소에의한장중첩증이많기때문에수술전에장경색의소견이없고환자의전신상태가양호하다면내시경적정복술을시도해볼수있다는보고도있다. 1,5-7 이에저자들은회결장형장중첩증이있는 2명의환자에서비록그유발병소를뚜렷하게확인할수는없었으나수술전대장내시경을시행하여장중첩증을진단하고수술전내시경정복을시도해본환자를경험하였기에보고한다. 증례 1. 증례 1 76세남자가한달전부터심해진간헐적인우하복부통증을주소로내원하였다. 환자는당뇨, 고혈압약물치료중이며 17년전위궤양에의한천공으로위부분절제술을받은병력이있었다. 내원당시혈압 130/80 mmhg, 293

294 대한소화기내시경학회지 2008;37:293-298 Figure 1. Abdominal CT findings. (A) It shows a target-like lesion in the right abdomen which is intussuscepting into the ascending colon. Also, about a 3.

Figure 2. Colonscopic finding. It shows a huge coil-spring reddish polypoid mass which is occupying two-thirds of the lumen of the ascending colon. 맥박수 78회 / 분, 체온 37.2 o C, 호흡수 20회 / 분이었다.

흉부및복부단순촬영에서특이소견은없었으며복부초음파와복부전산화단층촬영에서근위부상행결장부위에특징적인표적징후 (target sign) 와함께 3.3 cm 크기의종괴가관찰되었다 (Fig. 1A). 입원중환자의복부통증이심한상태가아니었고전신상태도양호한상태여서병변확인을위해장전처치를시행한후대장내시경검사를시행하였다.

2 294 대한소화기내시경학회지 2008;37: Figure 1. Abdominal CT findings. (A) It shows a target-like lesion in the right abdomen which is intussuscepting into the ascending colon. Also, about a 3.3 cm-sized cystic mass is observed in intussusceptum (arrow). (B) It shows sausage shaped intussusception including terminal ileum and ascending colon, extending into the distal transverse colon. Figure 2. Colonscopic finding. It shows a huge coil-spring reddish polypoid mass which is occupying two-thirds of the lumen of the ascending colon. 맥박수 78회 / 분, 체온 37.2 o C, 호흡수 20회 / 분이었다. 신체검사소견에서하복부에경한압통이있었으나종괴는촉지되지않았고간및비장비대는없었다. 말초혈액검사에서백혈구 7,990/mm 3, 혈색소 12.7 g/dl, 혈소판 413,000/mm 3 였고, 혈청생화학검사에서총단백 6.2 g/dl, 알부민 3.7 g/dl, AST/ALT 12/8 IU/L, 총빌리루빈 0.5 mg/dl, BUN/Cr 23/0.8 mg/dl였다. 흉부및복부단순촬영에서특이소견은없었으며복부초음파와복부전산화단층촬영에서근위부상행결장부위에특징적인표적징후 (target sign) 와함께 3.3 cm 크기의종괴가관찰되었다 (Fig. 1A). 입원중환자의복부통증이심한상태가아니었고전신상태도양호한상태여서병변확인을위해장전처치를시행한후대장내시경검사를시행하였다. 내시경으로관찰했을때간만 곡부위에항문쪽을항해내강가운데로돌출되어있는종괴를관찰할수있었으나유발병변을뚜렷하게확인할수는없었다. 공기를주입하면서관찰했을때상행결장내에허혈성손상은없는상태였으며감입부가구강쪽방향인상행결장내로밀려올라가면서회장이결장내로중첩된모양을관찰할수있었다 (Fig. 2). 회맹판은관찰할수없었으며중첩부위의가운데로내시경을진입시키려고했으나저항감이심하여실패하였고, 악성종양에의한장중첩증가능성이높아개복수술을시행하였다. 수술전에시행한양전자방출단층촬영 (PET/CT) 에서도악성종양이의심되었다. 수술소견에서우측결장과맹장에회장이약 15 cm 가량말려들어가있었고장중첩증원위부장관의확장및장관벽의부종을보여우반결장절제술을시행하였다. 절제한회장의길이는 25 cm였고절개했을때회맹판에서 14 cm 상방에 cm의난원형종괴가내강내로돌출되어있었다 (Fig. 3A). 조직검사소견에서병변은점막하층에위치하였고점액성간질내에방추세포, 혈관, 호산구로생각되는염증세포의침윤이관찰되었다 (Fig. 3B). 면역조직화학염색결과 c-kit 음성소견을보였으며조직검사결과염증성섬유모양용종으로확인되었다. 환자는수술후 15일째합병증없이퇴원하여현재외래추적관찰중이다. 2. 증례 2 51세남자가 1달전부터시작된간헐적인하복부통증을주소로내원하였다. 내원당시혈압 150/90 mmhg, 맥박수 70회 / 분, 체온 37.2 o C, 호흡수 20회 / 분이었으며, 신체검사에서하복부에경한압통이있었으나종괴는촉지되지않았고간및비장비대는없었다. 말초혈액검사에서백혈구 14,000/mm 3, 혈색소 13.7 g/dl, 혈소판 450,000/mm 3 였고, 혈청생화학검사에서총단백 6.1 g/dl,

이일영외 : 수술전대장내시경정복을시도한회결장형장중첩증 2 예 295 Figure 3. Pathologic findings.

ascending colon. (B) The polyp is composed of proliferated fibroblasts with collagen deposition and numerous eosinophils, microscopically (H&E stain, 400). Figure 4. Fluoroscopic findings.

(B) Also, colonoscope is advanced to the mildly dilated distal ileum but intussusception was not reduced. 알부민 3.7 g/dl, AST/ALT 15/8 IU/L, 총빌리루빈 0.4 mg/dl, BUN/Cr 10/0.7 mg/dl였다.

첫번째증례환자의경우처럼전신상태를살펴볼때장경색소견은없었으나장전처치에는어려움이있어관장을시행한후대장내시경을시행하였다. 횡행결장중간부위를넘어서면서내강가운데로돌출되어있는감입부를관찰할수있었고횡행결장에는장허혈이나경색을의심할만한점막변화는없는상태였다.

3 이일영외 : 수술전대장내시경정복을시도한회결장형장중첩증 2 예 295 Figure 3. Pathologic findings. (A) On opening the colon, the intussceptum of ileum, measuring 14 cm in length, a 3 cm-sized pedunculated ovoid polyp at the tip of the resected specimen (arrow) is found in the lumen of ascending colon. (B) The polyp is composed of proliferated fibroblasts with collagen deposition and numerous eosinophils, microscopically (H&E stain, 400). Figure 4. Fluoroscopic findings. (A) Colonoscope is reached to the terminal ileum by air insufflation and gastrographin injection. (B) Also, colonoscope is advanced to the mildly dilated distal ileum but intussusception was not reduced. 알부민 3.7 g/dl, AST/ALT 15/8 IU/L, 총빌리루빈 0.4 mg/dl, BUN/Cr 10/0.7 mg/dl였다. 복부단순촬영에서는다발성공기액체층이관찰되었으며, 복부전산화단층촬영에서소세지모양의감입부가있었으며장중첩부위는말단회장과상행결장과함께횡행결장의말단부까지포함하고있었다 (Fig. 1B). 감입부내에뚜렷한유발병변은보이지않았으며감입부말단부위에비균질조영증강소견만관찰되었다. 첫번째증례환자의경우처럼전신상태를살펴볼때장경색소견은없었으나장전처치에는어려움이있어관장을시행한후대장내시경을시행하였다. 횡행결장중간부위를넘어서면서내강가운데로돌출되어있는감입부를관찰할수있었고횡행결장에는장허혈이나경색을의심할만한점막변화는없는상태였다. 회맹판을정확히관찰할수는없었지만중앙부위를따라첫번째증례의환자처럼조심스럽게내시경을진행했을때처음에는저항감이있어진입할수없었으나공기와수용성조영 제를소량주입한후좁아져있는장관의입구가열리는것을확인할수있었으며내시경으로전진해들어갔을때말단회장부위로진입할수있었다 (Fig. 4A). 삽입된원위부회장은경도로확장되어있었으며경증의부종이있는상태로좁아져있었으나점막손상소견은없는상태였다. 또한구강방향으로 20 cm 정도올라가면서관찰했을때유발병소로판단되는병변은발견할수없었다 (Fig. 4B). 환자는장중첩이심하여내시경적정복은어려울것으로판단하여개복술을시행하였다. 수술소견에서회결장형의장중첩증이관찰되었고, 회맹판은확인할수없는상태에서장중첩증원위부장관의확장및장관벽의부종을보여우반결장절제술을시행하였다. 절제한회장의길이는 5 cm였고결장의길이는 16 cm였다. 절개한회맹판부위에감입부가있었고삼출물에덮힌 cm 크기의악성종괴가관찰되었다 (Fig. 5A). 수술로얻은조직의종괴는회맹판에위치하고있었고 (Fig. 5B), 비전형적원주세포로이루어져

296 대한소화기내시경학회지 2008;37:293-298 Figure 5. Pathologic findings. (A) On opening, the part of distal ileum and ileocecal valve is protruded into the lumen of cecum.

(B) Gross section shows a white-gray fibrotic tumor on the cecal side of the ileocecal valve without a involvement in the terminal ileum (arrow).

있어과염색성핵과고도의핵세포질비를특징으로하는회맹판에서발생한대장암으로진단하였다 (Fig. 5C). 환자는수술후 12일째합병증없이퇴원하여현재외래추적관찰중이다. 고 장중첩증은주로소아에서흔히발생하며뚜렷한원인이없이발생하는경우가대부분이다.

4 296 대한소화기내시경학회지 2008;37: Figure 5. Pathologic findings. (A) On opening, the part of distal ileum and ileocecal valve is protruded into the lumen of cecum. The head of intussuceptum shows a white gray fungating mass covered by green exudate, measuring cm. (B) Gross section shows a white-gray fibrotic tumor on the cecal side of the ileocecal valve without a involvement in the terminal ileum (arrow). Cancer invades into the serosa and pericolic fat tissue without lymph node metastasis. (C) It shows moderately differentiated adenocarcinoma (H&E stain, 400). 있어과염색성핵과고도의핵세포질비를특징으로하는회맹판에서발생한대장암으로진단하였다 (Fig. 5C). 환자는수술후 12일째합병증없이퇴원하여현재외래추적관찰중이다. 고 장중첩증은주로소아에서흔히발생하며뚜렷한원인이없이발생하는경우가대부분이다. 이에반해성인에서는드물게발생하며 70 90% 이상에서그원인을찾을수있고, 악성종양이전체원인의 70% 정도로가장많으며특히 60세이상의고령에서많이생기는것으로보고되었다. 1,2 대개대장의장중첩증은악성종양때문이며소장에서는메켈게실이나양성종양에의한경우가가장많다. 8 Azar 등 2 은장폐색으로나타나는증상이가장흔히나타나고그외에체중감소, 발열, 변비, 설사, 복부종괴등으로나타날수있으며수일에서수년동안반복적으로간헐적인복통만호소할수도있다고보고하였다. 장중첩증의진단에있어서혈청학적검사소견은큰도움이안되며복부초음파및복부전산화단층촬영이확진에큰도움을주고있다. 4 본증례둘다복부전산화단층촬영에서표적징후가관찰되는회결장형장중첩증소견을보여진단에어려움은없었으나유발병소확인은어려웠다. 특히첫번째증례에서확인된염증성섬유모양용종은매우드문양성질환으로대부분위에서발생 찰 하고회장에서발생하는경우는대부분증상을동반하여장중첩증이나장폐색을유발하는것으로알려져있다 대부분의성인장중첩증환자는장기간의간헐적이고부분적인장폐쇄증상으로내원하는데이는성인의경우소아에비해장관의직경이커서장간막의압축이심하지않고림프관및정맥폐쇄가늦게오므로완전폐쇄보다는부분적인장폐쇄가많기때문이다. 8 성인장중첩증의치료방법에는현재대부분의보고들에서수술을원칙으로생각하고있으나뚜렷하게정립되어있는수술방법은아직없다. 1 또한중첩된장을절제하기전에먼저도수정복을하는것에대해서도논란이있는데, 소장과우측결장의경우먼저도수정복을시도하여유발병소를확인한다음절제를하고이미장허혈, 장경색으로손상되어살릴수없는소장이나횡행결장, 하행결장에장중첩증이발생한경우는내시경정복없이수술적절제를한다. 이는소장의경우양성유발병소가많고좌측결장의경우는악성의빈도가높기때문이며내시경정복시무리한조작으로천공이나악성종양세포의파종이우려될경우에는정복없이광범위한수술적절제를하도록권유되고있다. 1,7,13,14 그러나, 최근일부의환자이지만장중첩증을대장내시경으로정복한예가있으며 5,6 특히회결장형장중첩증의경우양성질환에의한경우가많아부종이나허혈이동반된장경색의경우가아니라면수술전에대장내시경을시행하여유발병변을관찰할수있고이때주입되는일정량의공기에의해서도정복되는경우가있을수있기

5 이일영외 : 수술전대장내시경정복을시도한회결장형장중첩증 2 예 297 때문에수술전대장내시경시행이도움이된다는보고가있다. 13 그러나, 수술전대장내시경검사시돌출종괴에대한조직검사는되도록이면시행하지말아야하는데그이유는조직검사자체가장중첩이된장관의혈액순환에장애를주어조직괴사를조장할가능성이있기때문이며또한유발병소가아닌곳의조직검사는의미가없기때문이다. 14 또한비록수술을시행하더라도회결장형장중첩증의경우중첩된회장이매우긴경우가많아정복을먼저시도해본다면과도한절제를막는데도움을줄수있을것이다. 즉, 우측결장을포함하고있으면원인이되는유발부위가회장, 회맹부혹은대장인가를구분하기가쉽지않아불필요한확대절제가시행될수도있다는것이다. 1,8,15 본증례들중첫번째환자의경우는환자의연령이 60세이상이었으나유발병소가염증성섬유모양용종으로보고되어특히내시경적정복또는절제전수술장에서의정복이필요한경우라고할수있다. 본증례들에서시행한수술전대장내시경검사는대장병변의심시시행은할수있으나폐쇄성종괴를확인할뿐장중첩증을진단하기는부적절하다는보고도있으나증례두번째환자처럼감입부를지나서말단회장내로조영제를주입하면서관찰한다면내시경적으로장중첩증을진단할수있고대장조영술을대치할진단술기로도효용성이있다고할수있을것이다. 또한본증례들에서는정복되지는않았지만내시경을통한공기주입으로중첩된장을정복하여진단및치료한예, 바륨조영술검사에서이상소견으로관찰된회장점막탈출 (prolapse) 을내시경을이용한공기주입으로정복한예, 그리고수술전동시성종양의발견에중요한검사법이될수있다는점을고려하면수술전대장내시경검사로인한합병증의우려보다는장점이더많아장중첩증이의심될때는대장내시경검사의시행을적극적으로고려해야한다고생각한다. 5,6,13 저자들은양성및악성병변에의해장중첩증이발생한성인환자에서비록내시경적완전정복은하지못했으나수술전대장내시경으로장중첩증을진단하고내시경적정복을시도해본환자를경험하였기에문헌고찰과함께보고한다. ABSTRACT In contrast to the idiopathic cause of intussusception in children, adult intussusception in most patients is associated with organic causes. The majority of these patients are brought to the operating room with the preoperative diagnosis of bowel obstruction, and the surgeon discovers an intussusception intraoperatively. But the increasing use of abdominal CT may improve the ability to diagnose intussusception. There is no universal agreement upon the correct treatment of adult intussusception, although most authors agree that surgical intervention is necessary. In the more recent reports, colonoscopic reduction of intussusception has been reported for selected patients. For patients in whom the involved ileum is extremely long, it is advisable to attempt an operative reduction or colonoscopic reduction selectively. Thus, we report here on two patients with benign and malignant lesion, respectively, that caused ileocolic intussusception; preoperative colonoscopic diagnosis and reduction were attempted for these patients, although the patients were not reduced by colonoscopic procedure. (Korean J Gastrointest Endosc 2008;37: ) Key Words: Intussusception, Surgery, Ileocolic, Colonoscopy, Reduction 참고문헌 1. Omori H, Asahi H, Inoue Y, Irinoda T, Takahashi M, Saito K. Intussusception in adults: a 21-year experience in the university-affiliated emergency center and indication for nonoperative reduction. Dig Surg 2003;20: Azar T, Berger DL. Adult intussusception. Ann Surg 1997; 226: Nagorney DM, Sarr MG, McIlrath DC. Surgical management of intussusception in the adult. Ann Surg 1981;193: Reijnen HA, Joosten HJ, de Boer HH. Diagnosis and treatment of adult intussusception. Am J Surg 1989;158: Kim KM, Kim JH, Ha TI, et al. A case of adult idiopathic ileocecal intussusception reduced by colonoscopy. Korean J Gastrointest Endosc 2005;30: Eu KW, Seow C, Goh HS. Caecal mass on barium enema study-a case for routine colonoscopy. Singapore Med J 1994; 35: Chae GR, Cheon HD, Tae HJ, Kim CS, Lee KM, Ju MJ. Two cases of adult intussusception. J Korean Soc Coloproctol 2001; 17: Kim DH, Chae GB, Choi WJ, Song TJ, Choi SY, Moon HY. Diagnosis and management of adult intussusception. J Korean Surg Soc 1998;55: Lee CS, Nam SY, Park RY, et al. A case of adult intussusception caused by an inflammatory fibroid polyp. Korean J Gastroenterol 2002;40:64-67.

6 298 대한소화기내시경학회지 2008;37: Jabar MF, Prasannan S, Gul YA. Adult intussusception secondary to inflammatory polyps. Asian J Surg 2005;28: Bays D, Anagnostopoulos GK, Katsaounos E, Filis P, Missas S. Inflammatory fibroid polyp of the small intestine causing intussusception: a report of two cases. Dig Dis Sci 2004; 49: Park SY, Kang HG, Lee HJ, et al. A case of inflammatory fibroid polyp of the cecum causing intussusception. Korean J Gastrointest Endosc 2005;30: Park JH, Lee SH, Kim BG, et al. A case of cecal colon cancer ausing intussusception and synchronous sigmoid colon cancer. Korean J Gastrointest Endosc 2006;32: Chang FY, Cheng JT, Lai KH. Colonoscopic diagnosis of ileocolic intussusception in an adult. a case report. S Afr Med J 1990;77: Park YS, Lee JD, Seo YS, et al. A case of adenocarcinoma in ileocecal valve mimicking inflammatory bowel disease. Korean J Gastrointest Endosc 2002;25:

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Jkss hwp 급성복증을보이는감염성요막관기형 Infected Urachal Remnants with Symtoms of the Acute Abdomen: The Differential Diagnosis & Proper Management Jan Dy Lee, M.D., Chee Young Lim, M.D. 1, Hyoung Il Kim, M.D. 1, Chul Woon Chung,