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1 대한내과학회지 : 제 76 권제 4 호 2009 복통으로발견된대장과장간막의지방종증및장간막염전 1 예 가톨릭대학교의과대학내과학교실 이세민 정대영 고명범 노치호우성용 김진일 김재광 case of colonic and omental lipomatosis and omental torsion presenting with abdominal pain Se Min Lee, M.D., Dae Young Cheung, M.D, Myoung eom Koh, M.D., Chee Ho Noh, M.D., Seong Yong Woo, M.D., Jin Il Kim, M.D. and Jae Kwang Kim, M.D. Department of Internal Medicine, College of Medicine, The Catholic University of Korea, Seoul, Korea lipoma, one of the most commonly encountered submucosal tumors in the gastrointestinal tract, usually presents as one or a few lesions. Lipomatous polyposis, which is defined as the presence of multiple lipomas in the intestinal wall, is rare. Here, we report a case of colonic lipomatous polyposis that involved not only the colon, but also the omentum and skeletal muscle. The patient presented with right lower quadrant abdominal pain and was diagnosed using colonoscopy and computed tomography (CT). The abdominal pain caused by omental torsion due to an omental lipoma resolved after conservative treatment without surgical intervention. (Korean J Med 76: , 2009) Key Words: Lipomatous polyposis; Omental torsion; Generalized lipomatosis 서론지방종은과형성성용종과선종성용종에이어대장에서세번째로흔하게발생하는양성종양으로보통단발성으로발생하며 4개이상발견되는경우는드물다 1). 특히다발성으로 1 cm 이하의지방종들이무수히대장에서발견되는경우지방종성용종증이라하며비유전성대장용종증의하나로매우드물어 1-4), 외국에몇예만이보고되어있고국내에는보고된바가없다. 외국에발표된증례도대장에만지방종증 이있거나복막에지방종을동반하여소장의염전으로발견된경우 5) 만이보고되어있으나저자들은장간막의지방종으로인해이차적으로장간막염전이온급성복통환자에서우연히발견된지방종성용종증과근육간, 근육내, 피하의지방비후가같이동반된증례를경험하였기에보고한다. 증례환자 : 여자, 33세주소 : 내원전일부터발생된우하복부통증 Received: ccepted: Correspondence to Dae Young Cheung, M.D., St. Mary s Hospital, 62 Yeouido-dong, Yongdeungpo-gu, Seoul , Korea adagio@catholic.ac.kr

- The Korean Journal of Medicine: Vol. 76, No. 4, 2009 - C D Figure 1.

the right lower quadrant surrounded by adjacent whirls caused by increased fatty strands (solid arrow,,, D). C D Figure 2.

2 - The Korean Journal of Medicine: Vol. 76, No. 4, C D Figure 1. Enhanced abdominal CT shows a solitary large non-enhancing, fat-attenuated omental mass (arrowhead, C) with heterogeneous attenuation, which was presumed to be a lipoma (not confirmed surgically), in the right lower quadrant surrounded by adjacent whirls caused by increased fatty strands (solid arrow,,, D). C D Figure 2. Fat-attenuated nodular lesions involved the submucosal and subserosal layers of the entire colon (arrowhead,, ). Marked proliferative omental thickening (-D) and perirectal fat were demonstrated (double arrows, C, D). In addition, abdominal CT showed intramuscular lipomas within the back muscles bilaterally (double arrows, ) and huge intermuscular lipoma in both gluteal regions (solid arrow, C, D). 현병력 : 환자는내원전일오후갑자기우하복부통증이발생된뒤내원당일오전점점심해져내원하였다. 과거력 : 특이소견없음. 가족력 : 특이소견없음. 진찰소견 : 내원당시혈압은 120/80 mmhg, 맥박수 102회 / 분, 호흡수 20회 / 분, 체온은 36.7 로비교적안정적이었다. 환자는 155 cm에 61 kg, MI 25로약간과체중이었고, 외관 상특별한이상없었다. 이학적검사에서복부는과체중으로인해지방조직이두꺼웠으나촉진에서부드러웠고우하복부에약한압통이있었으나반발통은없었으며정상장음이청진되었다. 검사실소견 : 일반혈액검사에서백혈구 6850/mm 3, 혈색소 12.0 g/dl, 혈소판 250,000/mm 3, 중성구 64.8% 였으며 ST 15 IU/L, LT 11 IU/L, LP 361 IU/L, 총빌리루빈

Figure 4. Microscopic examination of the submucosal colonic tumor confirmed its lipomatous nature and the submucosal position of the polyps (left: H&E stain, 100, right: H&E stain, 400).

3 - Se Min Lee, et al. Case of generalized lipomatosis presenting with abdominal Pain - C Figure 3. Colonoscopy showed multiple diverticuli of the ascending, transverse, descending, and sigmoid colon (), and numerous small round yellowish submucosal tumors in the sigmoid colon and rectum (, C). Figure 4. Microscopic examination of the submucosal colonic tumor confirmed its lipomatous nature and the submucosal position of the polyps (left: H&E stain, 100, right: H&E stain, 400). mg/dl, Na 144 meq/l, K 4.2 meq/l, amylase 110 IU/L, 총콜레스테롤 137 mg/dl, 중성지방 28 mg/dl, 고밀도지단백 56 mg/dl, 총지질 475 mg/dl로 LP가약간상승되어있는것외에다른이상은발견되지않았다. 방사선학적검사 : 단순흉부 X-선촬영은정상이었고, 복부단순촬영에서우상복부에기능성장폐쇄로인한약간의공기가관찰되었다. 치료및경과 : 환자는지속적으로우하복부의통증을호소하고있었으므로충수돌기염을의심하여시행한복부컴퓨터단층촬영에서우하복부장간막에지방음영을보이는원형의종괴와주변의장간막이소용돌이모양으로염전되어있었으며 ( 그림 1), 대장벽에미만성지방음영 ( 그림 2, 2), 양쪽대퇴부근육내의두꺼운지방층, 상체의피하지방층의비후와직장주변지방의비후로자궁이앞쪽으로밀려있는것 ( 그림 2C, 2D) 이관찰되었다. 우하복부의장간막염전이복통의원인으로생각되었으나사진에서염전된부위에경색을의심할만한소견이없고발열이없으며혈액검사에서도백혈구증가증등의큰이상이없었으므로수술적치 료보다는일단금식후항생제를투여하면서관찰하기로하였다. 장간막염전의선행요인이된원형의종괴가지방음영을보이고있었으므로종괴는지방종으로생각하였고, 대장벽에미만성지방음영이있었으므로자세히관찰하기위하여대장내시경을시행하였다. 대장내시경에서상행결장부터구불창자까지여러개의게실이, 구불창자부터직장까지는작고둥근황색조의점막하종양이무수히관찰되었다 ( 그림 3). 황색조의점막하종양은작은지방종으로생각되었고, 확진을위하여조직생검을시행, 점막하층에두꺼운지방층을관찰하였다 ( 그림 4). 위의소견을종합하여전신의지방종증으로진단하였고, 환자는대증적치료 10일뒤증상호전되어퇴원하였다. 고찰장간막염전은일차성이나이차성으로장간막이염전되어복통을일으키고심하면경색이일어나조직괴사에이르게하는질환으로급성복통의원인중매우드문것으로되어있다 6,7). 이분성장간막, 부장간막 (accessory omentum),

4 - 대한내과학회지 : 제 76 권제 4 호통권제 584 호 좁은장간막경 (omental pedicle), 수술후장유착등해부학적이상이있거나비만할때이차성으로나타나며일차성장간막염전은매우드물기때문에임상에서흔하게접하지않아특별한과거력이없는급성복통환자를접하였을때쉽게생각할수없다 8). 그뿐아니라보통좌측부위보다우측부위의장간막이더쉽게움직일수있고크기때문에염전이쉽게일어나우하복부통증으로나타나기쉬우며, 복부전산화단층촬영에서특유의소용돌이장간막모양이나타나지않으면충수돌기염으로오인하여수술장에서진단을내리는경우가많다 6). 점막하종양인지방종은위장관에흔하게나타나며특히대장에서단발성또는 4개이하로흔하게발생하나 1), 4개이상다발성특히용종증으로나타나는경우는매우드물다 1-3). 보통의지방종은위치와크기에따라증상을일으키는데 9,10) 1 cm 미만의지방종은거의증상이없으며 4 cm 이상은 75% 이상에서증상을일으킨다 9). 지방종성용종증은 2~5 mm의매우작은지방종이대장에무수히있는것으로크기가작아보통증상이없으며악성변화도일으키지않아 2), 우연히발견되는경우가대부분이나드물게위장관출혈, 설사, 장중첩증또는장폐쇄등이나타날수있으며본증례에서와같이보통은매우경미하다 4). 지방종성용종증은모든연령에서발생될수있으나주로 40~50대에발견되는경우가많고성별분포도는밝혀진바가없다 2). 보통의지방종은선종성용종과는반대로맹장이나회맹판, 상행결장, 구불창자순으로많이생기지만지방종성용종증은미만성으로소장또는대장전체에나타나는경우가많고 4,5). 그중에서도하행결장과구불창자에특히많은수의용종이몰려서나타난다 1,11). 보통게실은결장끈의구조적변화와대장내강내의압력변화로인하여대장벽의약한부위가밖으로돌출되는것으로나이에따른변화라할수있는데, 지방종성용종증은지방종에의해약해진부분의점막층과점막근판이고유근층을뚫고쉽게돌출되므로게실이동반되는경우가많다 3). 본증례에선지방종이없는곳에게실이있었고, 지방종이있는곳은게실이관찰되지않았으므로나이에따른변화로인한게실로생각할수있다. 복막의지방종증이지방종성용종증과피하의지방조직비후와동반되어나타난 1예 2) 가보고되어있으나여러지방종증을동반한환자에서장간막염전이더잘일어난다는보고는없으며그런증례도현재까지보고된바가없다. 본증례에서는복부전산화단층촬영에서보인장간막의큰지방종이선행요인이되어장간막염전이일어난것으로생각되나이환자가전신의 지방조직이비후된점을볼때사진에서보이는지방종외에도염전된장간막주위에영상학적검사에서보이지않는작은지방종들이있고장간막의지방조직자체가매우비후되어고정점으로작용하면서장간막염전이쉽게일어났을것으로생각할수있으나전신적지방종증자체가매우드물어이로인한장간막염전의증례나장간막염전의가능성에대한보고가없는것으로생각되며이에대한좀더많은증례보고와고찰이필요하겠다. 지방종성용종증이비만과연관이있다는보고는현재까지없으며 Santos 등 1) 이발표한증례의환자는비만이었다고기술되어있으나다른증례에서는특별한언급이없었고, 비만과의특별한연관성은알려진바가없다. 피부과영역에서보고된유전질환인가족성다발성용종증도피부에는수많은지방종이있지만다른기관의지방종은없었다 13). 본증례는피부에눈에보이는지방종은없으나장간막지방종, 지방종성용종증과복강내의여러지방조직의비후외에도근육내와근육간의지방비후, 상체의피하지방비후등전신의지방비후를동반하고있다. 이런전신의지방비후환자에서지방조직이계속늘어나는지, 계속늘어난다면향후심혈관계에어떤영향을미치며어떤문제를일으킬수있는지, 치료가가능한지등은추적관찰하면서주시해야할점이다. 장간막염전은원칙적으로선행요인을해결하고염전되어경색된부위를절제하여야한다 6-8,12). 최근에는비수술적치료가성공적으로시도되고있으나 14), 복강내에괴사된조직을남겨농양을형성할여지가있다 15). 본증례도대증치료로증상이좋아졌으나장간막의지방종은계속남아다시염전을일으킬가능성이충분히있으므로증상이재발한다면근본적인수술적치료로선행요인인지방종제거를고려해야하겠다. 저자들은현재까지보고된바가없는전신의지방비후환자를접하였기에보고하는바이며두가지이상의기관에지방종이발견된환자는다른기관의지방비후여부를주의깊게관찰하여향후일어날수있는문제를예측하고문제가발생했을경우적절하게대응할수있어야하겠다. 요약장간막염전은급성복통의드문원인으로해부학적구조에이상이있거나비만한경우등어떤선행요인이있을때이차성으로나타나지만일차성으로도나타날수있다. 대장은지방종이가장호발하는위장관으로모든위장관지방종의 60~70% 가발생하지만다발성으로발생하는지방종성용

5 - 이세민외 6 인. 복통으로발견된전신의지방종증 1 예 - 종증은매우드물어외국에만 8예정도가보고되어있다. 저자들은쉽게접하기어려운장간막염전환자에서지방종성용종증과근육내지방층의비후가동반되어있는증례를접하였다. 이와같은전신의지방종증은국내에알려진바가없으며외국에서현재까지알려진위장관의지방종증이나피부과영역에서알려져있는가족성다발성지방종증과도또다른지방종증이기에문헌고찰과함께보고한다. 중심단어 : 장간막염전 ; 지방종성용종증 ; 전신의지방비후 REFERENCES 1) Santos-riz, Garcia JP, Gonzalez C, Colina F. Lipomatous polyposis of colon. Histopathology 38:81-83, ) Ward EM, Wolfsen HC. Review article: the non-inherited gastrointestinal polyposis syndrome. liment Pharmacol Ther 16: , ) rouland JP, Poupard, Nemeth J, Valleur P. Lipomatous polyposis of the colon with multiple lipomas of peritoneal folds and giant diverticulosis: report of a case. Dis Colon Rectum 43: , ) Ramirez JM, Ortego J, Deus J, ustamante E, Lozano R, Dominguez M. Lipomatous polyposis of the colon. r J Surg 80: , ) Neilson D, Wilkinson N, Magell J. Case of simultaneous diverticulosis, lipomatosis and volvulus of the small intestine. r J Surg 77:105, ) Poujade O, Ghiles E, Senasli. Primary torsion of the greater omentum: case report review of literature: diagnosis cannot always be performed before surgery. Surg Laparosc Endosc Percutan Tech 17:54-55, ) Itinteang T, van Gelderen WF, Irwin RJ. Omental whirl: torsion of the greater omentum. NZ J Surg 74: , ) Zargar NU, Kundal K, Krishna. Omental infarction: an unrecognized cause of acute abdomen. Indian J Pediatr 74:87, ) Zografos G, Tsekouras DK, Lagoudianakis EE, Karantzikos G. Small intestinal lipoma as a cause of massive gastrointestinal bleeding identified by intraoperative enteroscopy: a case report and review of the literature. Dig Dis Sci 50: , ) Chung YF, Ho YH, Nyam DC, Leong F, Seow-Choen F. Management of colonic lipomas. ust N Z J Surg 68: , ) Reeder PH, Hopens T. Intestinal lipomatosis: an unusual case. m J Gastroenterol 78: , ) El Hajj II, Otrock ZK, Sharara I. Primary omental torsion: radiologic diagnosis in a young woman. Dig Dis Sci 50:1169, ) Gologorsky Y, Gologorsky D, Yarygina S, Surti U, Zirwas MJ. Familial multiple lipomatosis: report of a new family. Cutis 79: , ) Nagar H, Kessler, en-sira L, Klepikov I, Wiess J, Graif M. Omental infarction: an unusual cause of acute abdomen in children. Pediatr Surg Int 19: , ) Helmrath M, Dorfman SR, Minifee PK, loss RS, randt ML, Deakey ME. Right lower quadrant pain in children caused by omental infarction. m J Surg 182: ,

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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,