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1 대한내과학회지 : 제 76 권제 2 호 2009 등쪽췌장무발생과폐낭종성병변을동반한위의중복낭종 가톨릭대학교의과대학 1 내과학교실, 2 외과학교실 임성민 1 이혁 1 전은정 1 오정환 1 이상학 1 최상욱 1 이성 2 Gastric duplication cyst associated with dorsal pancreatic agenesis and a cystic pulmonary lesion Sung Min Lim, M.D. 1, Hyeug Lee, M.D. 1, Eun Jung Jeon, M.D. 1, Jung Hwan Oh, M.D. 1, Sang Haak Lee, M.D. 1, Sang Wook Choi, M.D. 1 and Seong Lee, M.D. 2 Departments of 1 Internal Medicine and 2 General Surgery, The Catholic University of Korea College of Medicine, Seoul, Korea Gastric duplication cysts and dorsal pancreatic agenesis are rare congenital anomalies, and little is known of the association between these two anomalies. A 17-year-old woman was admitted with a cystic lung mass detected as part of a health screening program. Chest computed tomography (CT) showed a cystic lung lesion in the right lower lobe and an incidental cystic abdominal mass. She had no specific symptoms or signs. Abdominal CT and endoscopic ultrasonography (EUS) revealed a cm cystic mass and agenesis of the body and tail of the pancreas. The cyst was removed successfully using laparoscopic gastric wedge resection. Microscopically, the muscle coat of the cyst wall was fused with the muscle layer of the stomach. Therefore, the cystic mass was diagnosed as a gastric duplication cyst. We present a patient with a pulmonary cystic lesion associated with two congenital anomalies: a gastric duplication cyst and dorsal pancreatic agenesis. (Korean J Med 76: , 2009) Key Words: Digestive system abnormalities; Duplication cyst; Stomach; Pancreas 서 론 과함께보고하는바이다. 위의중복 (gastric duplication) 은흔하지않은선천성기형으로, 약 70% 가 12세이전에진단되며, 성인에서는매우드물고, 특이한증상이나징후가없이발견되기도한다 1, 2). 췌장의무발생역시드문선천성기형으로당뇨병이나췌장염과같은합병증과동반하여보고되고있다 3). 저자들은건강검진중우연히발견된폐의낭종성병변으로내원하여위의중복낭종과등쪽췌장무발생이진단된예를문헌고찰 증례환자 : 강, 여자, 17세주소 : 만성기침현병력 : 환자는건강검진으로시행한흉부 X-선에서이상소견으로내원하였다. 과거력 : 당뇨병, 고혈압, 결핵, 간염등의과거력은없었으며, 수술력이나입원력없었다. 1년간하루 1갑흡연한병 Received: Accepted: Correspondence to: Jung Hwan Oh, M.D. Department of Internal Medicine, St. Paul's Hospital, The Catholic University of Korea, , Jeonnong 2-dong,Dongdaemun-gu, Seoul , Korea ojh@catholic.ac.kr

2 The Korean Journal of Medicine: Vol. 76, No. 2, 2009 A B Figure 1. (A) The chest radiograph shows cystic lung disease in the right lower lobe. (B) Computed tomography of the lung. Bifurcating tubular structures containing hypodense material and air-fluid levels are noted in the posterior basal segment of the right lower lobe. B A Figure 2. Contrast-enhanced computed tomography of the abdomen. (A) A cm cystic mass is noted in the left subphrenic space and is attached to the posterior of the gastric fundus and body. A few fold-like structures are seen within this lesion. (B) The body and tail of the pancreas are not visualized. 력 있었으며, 음주력은 없었다. 관찰되지 않았다. 3 가족력: 가족력에서 선천성 기형이나 다른 질환의 병력 은 없었다. 검사실 소견: 혈액 검사에서 백혈구 5,400/mm (호중구 50.8%, 림프구 39.7%, 호산구 1.9%), 혈색소 10.9 g/dl, 헤마 신체검사 소견: 내원시 혈압은 110/70 mmhg, 맥박은 분 3 토크리트 32.9%, 혈소판 274,000/mm, 프로트롬빈 시간 12.6 당 80회, 호흡수 분당 20회, 체온 36.5 이었고, 의식은 명료 초(96%), 활성화부분트롬보플라스틴 시간 30.9초이었고, 생 하였다. 흉부 검진에서 우하엽 부위에서 수포음을 동반한 화학 검사에서 혈청 AST 16 IU/L, ALT 10 IU/L, 총 단백질 거친 호흡음이 청진되었으며, 복부 검진에서는 특별한 소견 7.2 g/dl, 알부민 4.5 g/dl, 총 빌리루빈 0.3 mg/dl, r-gtp

3 - Sung Min Lim, et al. Gastric duplication cyst with dorsal pancreatic agenesis and pulmonary cyst - Figure 3. Esophagogastroduodenoscopy shows extrinsic compression at the posterior wall of the gastric fundus. U/L, ALP 218 U/L, 아밀라제 73 mg/dl, BUN 16.0 mg/dl, Cr 0.8 mg/dl, 나트륨 141 meq/l, 칼륨 3.8 meq/l이었다. CRP 0.10 mg/dl (0~0.3), CEA 0.33 ng/ml, CA U/mL 로정상범위에있었다. 방사선소견 : 내원하여시행한흉부 X-선에서우하엽에증가된음영이있었으며 ( 그림 1A), 내부에공기액체층이관찰되었다. 흉부 CT에서우하엽뒤바닥구역에서낭종성병변이관찰되었으며, 내부에저음영의물질과공기층을포함하고있었다 ( 그림 1B). 또한상복부에위와비장사이에낭종성덩어리가관찰되었으며, 내부에는중격이형성되어있었다. 치료및경과 : 폐우하엽에관찰되는소견은낭종성결핵병변혹은점액성박힘 (mucoid impaction) 을동반한기관지확장증, 기관지폐쇄증등을감별해야할것으로생각되었으며, 객담검사, 기관지내시경검사및기관지세척을시행하였다. 기관지내시경검사에서우하엽뒤바닥구역기관지가외인성압박으로좁아져보이는것외에특별한기관지내의병변은없었고, 세균, 진균검사에서동정된균은없었으며, 항산균도말검사음성, 결핵균중합효소연쇄반응음성이었고, 세포검사에서악성세포는관찰되지않았다. 복부 CT 검사에서 cm 크기의낭종성덩어리가왼쪽횡경막하공간, 위바닥과위몸통뒤에서관찰되었으며, 내부에는몇개의주름같은구조물이관찰되었다. 낭종성덩어리와인접한위벽은얇아져있었다 ( 그림 2A). 췌장몸통과꼬리가관찰되지않아, 췌장몸통과꼬리의무발생 (agenesis) 이 Figure 4. Endoscopic ultrasonographic images. A large homogenous low echogenic cystic mass adjacent to the gastric muscle layer lies in the space just anterior to the splenic vein, between the left kidney and spleen, where the body and tail of the pancreas are usually located. The body and tail portions of the pancreas are not observed. Figure 5. Photomicrograph of the gastric duplication cyst. The cyst wall consisted of gastric foveolar epithelium, gastric glands, and subjacent bundles of smooth muscle (H&E stain 200). 의심되었다 ( 그림 2B). 위내시경검사에서위바닥뒷벽에서덩어리에의한외인성압박관찰되었다 ( 그림 3). 내시경초음파검사에서위바닥에탐색자를두고관찰하였을때, 위근층에붙어있는양상의저음영의균일한덩어리가관찰되었고, 비장이나좌측신장과의경계는명확하였으며, 췌장의몸통

4 - 대한내과학회지 : 제 76 권제 2 호통권제 582 호 과꼬리가관찰되지않고, 췌장의몸통과꼬리가위치되어야할공간에덩어리가위치하였다 ( 그림 4). 낭종성덩어리는위의중복낭종으로생각되었고, 위의중복낭종에서발생한출혈, 천공등의합병증과악성종양으로발전할가능성이있어수술적치료를시행하였다. 복강경을통하여위바닥뒤쪽에서 9 7 cm 크기의낭종성덩어리가관찰되었으며, 위에인접하여있었고, 횡경막이나췌장등주변조직과연결은관찰되지않았다. 낭종내부에는우유빛의분비물을포함하고있었으며, 복강경하위쐐기절제술을통하여낭종성덩어리는완전히절제되었다. 조직학적소견에서낭종은위조직에부착되어있었으며, 낭종내점막은위점막조직과같은양상이었고, 낭종벽은평활근층으로이루어져있어위의중복낭종으로확진할수있었다 ( 그림 5). 환자는수술후약 2개월간외래에서경과관찰중이며, 추적흉부 X선에서우하엽에관찰된낭종성병변은변화없는상태이다. 고찰위장관중복 (gastrointestinal duplication) 은혀에서항문까지위장관어디에서나발생할수있는선천성기형으로 4, 5), 중복의내측은위장관의점막, 중복의벽은평활근층으로구성되어, 인접위장관과교통성또는비교통성양상을보이는속이빈형태로관찰된다 5, 6). 회장말단부가가장흔한호발부위이며, 식도, 대장, 공장에서상대적으로위보다많이발생한다 4). 위의중복낭종은대만부위가가장흔하고, 후벽, 소만, 전벽, 분문순으로발생하며 2, 7), 여성이남성에비해 2배의빈도로나타난다고알려져있다 2). 위의중복은전체위장관중복의 2~8% 정도로드물며 6, 8), 1911년처음보고된이후로현재까지약 150개정도의보고가있다 9). 위의중복은관상혹은낭종형태로분류할수있는데 2, 6), 관상중복은드문형태이며위와내강이연결되어있는경우가있으나, 낭종중복은위벽일부분에인접하여있지만대부분에서위내강과직접적인연결은관찰되지않는다 2). 위의중복낭종의필수적인요소로낭종의벽은위벽과붙어있어야하며, 낭종은위근층의연장인평활근으로싸여있어야하고, 낭종의내벽은소화관의상피세포로덮혀있어야한다 10). 발생기전은정확하게알려져있지않으며여러가지가설로설명하고있다 2, 6). 관강의재소통이상 (errors of recanalization) 및세로주름의융합 (fusion of longitudinal folds) 에서발생한다는가설과척삭과배아내배엽의유착 (adhesion of notochord and embryonic endoderm) 에의해중복이형성된다 는가설, 발생학상의게실이지속된것 (persistent embryological diverticula) 이라는가설, 불완전쌍둥이형성 (abortive twinning), 저산소혹은손상과같은환경적요인등이주장되고있다. 임상증상은종괴의촉지, 구토와같이종괴에의한위장관폐쇄증상이흔하고, 낭종내부의위점막에서의궤양, 출혈, 천공등에의한복통, 체중감소, 빈혈, 흑색변, 토혈, 고열등이나타날수있고 2, 7), 위치와크기에따라다양하게나타난다. 약 20~50% 에서동반된기형을관찰할수있으며, 식도의중복이흔하고, 척추갈림 (cleft vertebra), 반척추뼈증 (hemivertebra), 척추융합과측만증 (vertebral fusion and scoliosis) 등척추의기형을동반하는경우가흔하다 5, 6). 또한위의중복낭종과함께반복적인췌장염이나다낭성신증과생식샘발생장애, 고가스트린혈증, 카르시노이드등이동반된경우도보고되고있다 8, 9). 중복낭종에서선암의발생도 6예정도알려져있으나 11), 중복낭종이진단되었을때악성잠재력이어느정도인지는아직불확실한상태이다 8, 9). 췌장무발생은드문선천성기형으로, 완전무발생이나배쪽무발생 (ventral agenesis) 은생존이어려운것으로알려져있으며 12), 등쪽무발생 (dorsal agenesis) 은드물게보고되고있다 3). 이전에는주로부검이나수술로진단하였지만, 최근 ERCP나 MRCP, 내시경초음파검사와같은영상진단검사를통한진단이이루어고있다 13). 췌장은십이지장으로발달될앞창자내배엽 (foregut endoderm) 에서배쪽과등쪽봉오리 (bud) 에서발생되며, 배쪽봉오리는췌장머리와갈고리돌기 (uncinate process) 를형성하며, 배쪽봉오리는몸통과꼬리를형성한다. 배쪽무발생은배쪽봉오리의형성부전으로인한것으로생각되고있다 3). 본증례에서는 CT와내시경초음파검사에서췌장몸통과꼬리의무발생이의심되었으며, 문헌고찰에서위의중복낭종과동반하여보고된증례는없었다. 앞창자 (foregut) 에서기원하는낭종은내층세포구성에따라장관낭종 (enteric cysts), 기관지낭종 (bronchogenic cysts), 식도중복낭종으로분류할수있다. 위와식도중복낭종의내층은대게소화관점막으로이루어지지만, 때로는기관지점막인거짓중층섬모원주상피 (pseudostratified ciliated columnar epithelium) 로구성되는경우가있어이런경우에는앞창자낭종이라부른다 6, 8, 9). 식도중복낭종과동반한기관지낭종, 폐분리증 (pulmonary sequestration), 기관지확장증, 기관지폐쇄증 (bronchial atresia) 등이보고되었으며 14), 위의중복낭종과폐분리증 15) 이동반된증례가보고되었다. Kitano 등 14)

5 - 임성민외 6 인. 등쪽췌장무발생과폐낭종을동반한위중복낭종 - 에의하면연결관이닫혀있는경우도있지만, 8명의증례모두에서식도중복낭종과폐의낭종성기형과의연결관이존재한다고하였다. 본증례에서는수술소견에서폐병변과의연결관은관찰할수없었고, 폐병변의직접적인수술적절제혹은조직학적검사가이루어지지않은상태라서연관성있는선천성기형인지여부에대한추가적인연구가필요할것으로생각한다. 이전에는수술전검사를통한감별이어려워중복낭종의진단이개복술로이루어지는경우가많았지만 1), 여러가지검사방법의발달로낭종의진단과감별에도움을받을수있게되었다 10, 16, 17). 상부위장관조영술에서내벽의종괴소견으로관찰되며, 복부초음파검사나 CT, MRI를통하여낭종벽과주변장기와의관계를확인할수있고, 췌장의가성낭종이나총담관낭종, 지라의낭종등과감별할수있다. 내시경초음파검사는위벽내병변과위벽외병변의감별에도움을줄수있으며 16), 중복낭종과평활근종이나지방종을감별할수있고, 초음파를통하여중복낭종에서저에코의근육층과고에코의점막층을구별할수도있다 10, 18). 내시경혹은경피적인낭종의흡인이진단및치료의목적으로시도되었지만 19), 출혈이나감염, 루형성 (fistula formation) 의위험성, 중복낭종의선암의가능성을고려하였을때, 낭종의흡인여부는신중히결정해야할것으로생각된다 10). 치료는절제하지않았을때합병증의가능성과선암의가능성을고려하여수술적치료가추천되고있다 1, 18) 내시경적올가미기법으로절제한증례도보고되었지만 20), 대게개복술혹은복강경을통하여절제하며, 수술적접근법의선택은아직논쟁의여지가남아있는상태이다 8, 17). 본증례에서는무증상이었지만, 여러가지합병증의가능성과선암의가능성을고려하여복강경을통한수술적치료를시행하여, 현재특별한합병증없이외래에서경과관찰중이다. 요약위의중복낭종은드문선천성기형으로, 본증례에서는특별한증상없이건강검진에서발견된폐의낭종성병변으로내원하여우연히진단되었다. 폐의낭종성기형과동반된식도중복이나위의중복은보고되고있으며, 환자의경우도연관된선천성기형가능성을배제할수없는상태이다. 또한특별한증상은없었지만, 검사에서우연히췌장의배쪽무발생이발견되었으며, 우리나라에서위의중복낭종과동반하여보고된증례는없다. 이에저자들은문헌고찰과함께증례를보고하는바이다. 중심단어 : 소화계이상 ; 중복낭종 ; 위 ; 췌장 REFERENCES 1) Perek A, Perek S, Kapan M, Goksoy E. Gastric duplication cyst. Dig Surg 17: , ) Wieczorek RL, Seidman I, Ranson JH, Ruoff M. Congenital duplication of the stomach: case report and review of the English literature. Am J Gastroenterol 79: , ) Fukuoka K, Ajiki T, Yamamoto M, Fujiwara H, Onoyama H, Fujita T, Katayama N, Mizuguchi K, Ikuta H, Kuroda Y, Hanioka K. Complete agenesis of the dorsal pancreas. J Hepatobiliary Pancreat Surg 6:94-97, ) Bower RJ, Sieber WK, Kiesewetter WB. Alimentary tract duplications in children. Ann Surg 188: , ) Stringer MD, Spitz L, Abel R, Kiely E, Drake DP, Agrawal M, Stark Y, Brereton RJ. Management of alimentary tract duplication in children. Br J Surg 82:74-78, ) Kim DH, Kim JS, Nam ES, Shin HS. Foregut duplication cyst of the stomach. Pathol Int 50: , ) Pruksapong C, Donovan RJ, Pinit A, Heldrich FJ. Gastric duplication. J Pediatr Surg 14:83-85, ) Wakabayashi H, Okano K, Yamamoto N, Suzuki Y, Inoue H, Kadota K, Haba R. Laparoscopically resected foregut duplication cyst (bronchogenic) of the stomach. Dig Dis Sci 52: , ) Cunningham SC, Hansel DE, Fishman EK, Cameron JL. Foregut duplication cyst of the stomach. J Gastrointest Surg 10: , ) Maeda H, Okabayashi T, Nishimori I, Kobayashi M, Morimoto K, Miyaji E, Kohsaki T, Hanazaki K, Onishi S. Diagnostic challenge to distinguish gastric duplication cyst from pancreatic cystic lesions in adult. Intern Med 46: , ) Kuraoka K, Nakayama H, Kagawa T, Ichikawa T, Yasui W. Adenocarcinoma arising from a gastric duplication cyst with invasion to the stomach: a case report with literature review. J Clin Pathol 57: , ) Voldsgaard P, Kryger-Baggesen N, Lisse I. Agenesis of pancreas. Acta Paediatr 83: , ) Sempere L, Aparicio JR, Martinez J, Casellas JA, de Madaria E, Perez-Mateo M. Role of endoscopic ultrasound in the diagnosis of agenesis of the dorsal pancreas. JOP 7: , ) Kitano Y, Iwanaka T, Tsuchida Y, Oka T. Esophageal duplication cyst associated with pulmonary cystic malformations. J Pediatr Surg 30: , ) Matsubayashi J, Ishida T, Ozawa T, Aoki T, Koyanagi Y, Mukai K. Subphrenic bronchopulmonary foregut malformation with pulmonary-sequestration-like features. Pathol Int 53: ,

6 - The Korean Journal of Medicine: Vol. 76, No. 2, ) Tanaka M, Akahoshi K, Chijiiwa Y, Sasaki I, Nawata H. Diagnostic value of endoscopic ultrasonography in an unusual case of gastric cyst. Am J Gastroenterol 90: , ) O'Donnell PL, Morrow JB, Fitzgerald TL. Adult gastric duplication cysts: a case report and review of literature. Am Surg 71: , ) Bottsford J, Meehan JJ. Common bile duct perforation resulting from a gastric duplication cyst. Pediatr Surg Int 24: , ) Ferrari AP Jr, van Dam J, Carr-Locke DL. Endoscopic needle aspiration of a gastric duplication cyst. Endoscopy 27: , ) Stecevic V, Karim R, Jacobs R. Gastric duplication cyst treated by endoscopic electrosurgical snare resection. Gastrointest Endosc 57: ,

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(49-54)Kjhps004.hwp Biliary Cystadenoma and Cystadenocarcinoma of the Liver Jae Hoon Lee, M.D., Dong Ho Choi, M.D. 1, Kyeong Geun Lee, M.D., Hwon Kyum Park M.D. and Kwang Soo Lee, M.D. Department of Surgery, College of Medicine,

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