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1 196 소아외과 : 제 16 권제 2 호 2010 년 Vol. 16, No. 2, December 2010 뇌실복강단락술후발생한복강내가성낭종 고려대학교의과대학외과학교실소아외과 부윤정 서 론 고문헌고찰과함께보고하는바이다. 복강내가성낭종 (Abdominal pseudocyst) 은뇌실복강단락술 (Ventriculoperitoneal shunt) 후발생할수있는흔하지않은합병증중하나로신경외과의사의시술후발생하는문제이나진단과치료에소아외과의사가관여해야하므로적절한진단과치료를알아둘필요가있다. 뇌실복강단락술은수두증 (hydrocephalus) 이있는환아에서치료목적으로널리시행되고있는방법으로주로소아신경외과전문의에의해시술되고있다. 이시술이점점활발하게시행되고환아의수명이연장되어감에따라그에따른합병증도늘어가고있는상태이다. 특히복강내에발생한합병증의경우소아외과의사와의협진이필수적으로요구되는바소아외과의사는이에대한증상및진단과치료에대해알아두어야하겠다. 본저자는소아에서뇌실복강단락술후발생한복강내가성낭종을경험하 접수일 : 10 / 11 / 16 게재승인일 : 10 / 12 / 24 교신저자 : 부윤정, 서울특별시성북구안암동 5 가고려대학교안암병원소아외과 Tel : 02) , Fax : 02) drboo@korea.ac.kr 증례 32개월된남아로복부팽만및복통을주소로내원하였다. 출생시조기진통으로 29주, 960 g에질식분만으로태어났으며수막척수탈출증 (meningomyelocele) 및 Chiari malformation type II, 이로인한수두증으로생후 3개월에수막척수탈출증복원술및복강뇌실단락술을시행하였다. 이후하반신마비있어재활치료중이었으며그외다른합병증없이잘지내던중 3일전부터발생한복부팽만및복부통증을주소로내원하였다. 복부진찰소견상중등도복부팽만이관찰되었으나복부는부드러웠고압통이나반발통은관찰되지않았다. 복부초음파를시행하였고초음파상 ( 그림 1) 우하복부에약 9cm 크기의내부에격막이있는거대한낭종이관찰되어뇌실복강단락술의합병증인가성낭종이의심되었으나타질환과의정확한감별을위해복부전산화단층촬영 (CT) 을시행하였다. CT상에서우하복부에주변장기와경계가뚜렷한장경 9cm 의두꺼운막으로형성되어있는낭종이관

찰되었고내부에뇌실복강단락의도관의끝이위치하고있는것으로보여가성낭종으로진단하였다 ( 그림 2). 검사를위해낭종내부액체를천자하여세균배양검사를시행하였고결과상균은검출되지않았다. 감염이없는뇌실단락술 도관원위부에발생한가성낭종으로진단하고수술을시행하였다.

2 부윤정 : 뇌실복강단락술후발생한복강내가성낭종 197 Fig. 1. Abdominal ultrasound howing huge cystic mass with internal septation on the right lower abdomen. Fig. 2. Abdomen CT, demonstrating a 9 cm-sized well defined cystic mass on the right side abdomen. In the cyst, tip of the shunt catheter is noticed. 찰되었고내부에뇌실복강단락의도관의끝이위치하고있는것으로보여가성낭종으로진단하였다 ( 그림 2). 검사를위해낭종내부액체를천자하여세균배양검사를시행하였고결과상균은검출되지않았다. 감염이없는뇌실단락술 도관원위부에발생한가성낭종으로진단하고수술을시행하였다. 수술소견상도관의끝에서부터발생한거대한낭종을관찰할수있었고내부의액체의성상은깨끗하였다 ( 그림 3). 가성낭종의일부가장과유착되어있고경계가모호하여낭벽의일부만절제하고광범위하게절개하여남아있는낭벽이충분히복강내로노출되도록하였다. 도관의원위부를새것으로교체한뒤끝을우측횡경막아래, 간위쪽으로꺾이지않도록주의하여위치시킨뒤아이의성장을고려하여충분한길이로남긴뒤고정하고수술을마쳤다. 외래에서시행한복부사진상도관의위치 ( 그림 4) 는꺾임이나다른문제없이잘기능하고있으며현재수술후 1년 6개월째로재발이나합병증없이양호한상태로외래추적관찰중이다.

198 소아외과제 16 권제 2 호 2010 년 Fig. 3. Operative findings. The wall of the pseudocyst is connected to the catheter tip (Left). Thick wall and contained clear liquid of the pseudocyst are visualized.

이러한단락술후발생하는합병증으로단락기능부전을일으키는경우는감염과카테터의폐쇄가대부분이며그중복강내발생하는합병증으로는도관의말단부가장, 배꼽, 방광, 질등을뚫고나오는경우 (extrusion), 탈장, 장천공, 장축염전이나장폐색증, 복수형성, 복막염, 음낭수종, 농양및가성낭종등이보고되었다 2,3.

3 198 소아외과제 16 권제 2 호 2010 년 Fig. 3. Operative findings. The wall of the pseudocyst is connected to the catheter tip (Left). Thick wall and contained clear liquid of the pseudocyst are visualized. Fig. 4. Plain abdominal x-ray, showing well-positioned VP shunt catheter, at 18 months after surgery. 고 찰 소아수두증에대한수술적치료로서의뇌실복강단락술은현재가장보편적으로이용되고있는방법으로최근에는복강경을 이용한수술법도보고되었다 1. 이러한단락술후발생하는합병증으로단락기능부전을일으키는경우는감염과카테터의폐쇄가대부분이며그중복강내발생하는합병증으로는도관의말단부가장, 배꼽, 방광, 질등을뚫고나오는경우 (extrusion), 탈장, 장천공, 장축염전이나장폐색증, 복수형성, 복막염, 음낭수종, 농양및가성낭종등이보고되었다 2,3. 뇌실복강단락술후에발생하는복강내가성낭종은 1954년 Harsh에의해처음보고된이래빈도는낮지만여러저자들에의해보고되어왔으나아직그원인과치료방법이확실치않은합병증으로알려져있다 4,5. 그발생빈도는정확하게측정하기어려우나 Anderson 등이발표한보고에따르면 0.7 % 4.5 % 에이른다고하며최근연구에의하면성인에비해소아에서단락술시행후합병증이더높게발생하는것으로보고되고있다 6,7. 가성낭종이란만성염증반응에의해발생한두꺼운섬유성막에의해형성된낭종을말하며다양한형태학적, 병리학적서술이

4 부윤정 : 뇌실복강단락술후발생한복강내가성낭종 199 존재하나원칙적으로중피세포로형성된내막 (mesothelial lining) 이없는경우가성낭종으로분류될수있다 6,8. 원인은다양하게제시되고있으나아직명백한원인은밝혀내지못한상태로이전의복부수술경력이나감염등이가장흔한원인으로보고되었다 6,9. 이는이전의반복적인수술이복강내유착을형성하고, 뇌척수액의흡수에장애를일으켜가성낭종을발생시킨다는가설로설명되고있으며, 외상이나수술로인한조직의손상및허혈, 그리고단락도관자체가이물염증반응을일으키게되면이또한가성낭종발생의원인이될수있다고한다 10,11. 이전에수술경력이없는환자에서도가성낭종이종종보고되고있으며그원인으로무증상감염을포함한도관의감염이가장유력하게제시되고있다. 그러나균동정검사상약 3분의 1정도에서만균이동정되는것으로보고되고있어원인에대한좀더심층적인연구가필요할것으로보인다 5. 복강내발생한가성낭종은우선적으로복부증상을보이게되는데이전의보고들에의하면대부분신경학적증상전에복부팽만, 복통등의복부증상이먼저나타나는것으로되어있다 8,12,13. 본증례에서도복부팽만이진행된후복부동통을호소하였고뇌신경증상은보이지않았다. 그러나소아에서는성인보다신경학적증상이동반되는경우가더흔하고보다빨리위험한상황에빠질수있으므로주의가필요하겠다 14. 진단은과거에는단순 X-ray 촬영이나단락조영술등이이용되기도했으나최근에는초음파나복부 CT 가많이이용되고있 으며다른질환과감별하는데에도용이하다 15. 진단을하는데있어가장중요한근거가되는소견은복강내액체음영을지닌경계가분명한낭종이관찰되고대부분내부에격막이없으며단락도관의끝이그내부에위치하고있는것이다. 이는그밖의다른복강내낭종성종괴나장간막낭종, 췌장의가성낭종등과감별이필요하다 16. 일단확진이되면신경학적증상이발생, 진행되기전에지체없이치료를시작해야하는데우선발열및백혈구증가등임상증상확인및천자를통해감염이있는지확인하는것이중요하다. 만약감염이있거나의심되는경우즉시도관을제거하거나도관의말단부를외부로배출시키고항생제치료를시작하여야한다 5. 이후에다시도관을복강내재진입시킬것인지에대해서는논란이있으며여러가지치료법이제시되고있다. 대부분의저자들이감염이없는경우낭종을제거하고도관을다시복강내재진입시키는데에동의하고있다 6,8,10,13. 본증례에서도수술전낭종천자를통해균동정검사및그람염색을시행하였으나감염소견이없는것이확인되어수술시도관말단부의복강내재거치를시행하였으며재발을방지하고안전한길이를확보하기위해우측횡경막아래, 간위쪽으로도관의위치를조정하여거치하였다. 그러나몇몇저자들은뇌실복강단락술을재실시한경우도관의감염여부와관계없이재발률이높다고보고하면서이를뇌실심방단락 (ventriculoatrial shunt), 뇌실요관단락 (ventriculoureteral shunt), 뇌실흉강단락 (ventriculopleural shunt), 혹은제삼뇌실조

5 200 소아외과제 16 권제 2 호 2010 년 루술 (third ventriculostomy) 등을시행해야한다고주장하고있다 5,17,18. 국내에서도허등 2 에의하면가성낭종이 4번이나재발하여결국뇌실요관단락술을시행하여호전이된경우를보고하였다. 하지만뇌실복강단락술이다른방법에비해합병증이적고안전한방법이므로감염이없고복강내상태가유착이나염증이심하지않고깨끗한경우복강내재거치를한번쯤재시도해보는것이더선호되고있다 6. 최근보고에의하면감염이없는경우복강경을이용하여낭종을배액하고크게절개하여연뒤도관을다른위치에재거치시키는방법을사용하여좋은결과를얻었다고하며단일통로복강경을이용한시술도보고되고있다 19,20. 뇌실단락술후발생하는복강내가성낭종은드물지만소아에서더흔히발생하는합병증으로아직까지효과적으로치료하는것이어려운질환이다. 본증례에서는가성낭종의부분적제거와복부내도관재거치로재발없이치료된경우로향후보다적절한치료방법에대한연구가필요하겠다. 참고문헌 1. Esposito C, Colella G, Settimi A, Centonze A, Signorelli F, Ascione G, Palmieri A, Gangemi M: One-trocar laparoscopy. Surgical Endoscopy 17: , Huh R, Shin WH, Lee K, Choi SK, Byun BJ, Kim ME, Moon C, Lee IS: Ventriculoureteral Shunt Operation for Treatment of Recurrent Abdominal Pseudocyst Complicating Ventriculoperitoneal Shunt: Case Report and Technical Note. J Korean Neurosurg Soc 22: , Bryant MS, Bremer AM, Tepas JJ, Mollitt DL, Nquyen TQ, Talbert JL: Abdominal complications of ventriculoperitoneal shunts: Case reports and review of the literature. American surgeon 54:50-55, Harsh GR, 3rd: Peritoneal shunt for hydrocephalus, utilizing the fimbria of the fallopian tube for entrance to the peritoneal cavity. J Neurosurg 11: , Mobley LW, 3rd, Doran SE, Hellbusch LC: Abdominal pseudocyst: predisposing factors and treatment algorithm. Pediatr Neurosurg 41:77-83, Anderson C: Intra-abdominal pseudocysts as a complication of ventriculoperitoneal shunts: a case report and review of the literature. Current Surgery 60: , Wu Y, Green N, Wrensch M, Zhao S, Gupta N: Ventriculoperitoneal shunt complications in California: 1990 to Neurosurgery 61: , Parry SW, Schuhmacher JF, Llewellyn RC: Abdominal pseudocysts and ascites formation after ventriculoperitoneal shunt procedures. Report of four cases. J Neurosurg 43: , Fischer EG, Shillito J, Jr.: Large abdominal cysts: a complication of peritoneal shunts. Report of three cases. J Neurosurg 31: , Latchaw JP, Jr., Hahn JF: Intraperitoneal pseudocyst associated with peritoneal shunt. Neurosurgery 8: , White B, Kropp K, Rayport M: Abdominal cerebrospinal fluid pseudocyst: occurrence after intraperitoneal urological surgery in children with ventriculoperitoneal shunts. J Urol 146: , 1991

6 부윤정 : 뇌실복강단락술후발생한복강내가성낭종 Grosfeld JL, Cooney DR, Smith J, Campbell RL: Intra-abdominal complications following ventriculoperitoneal shunt procedures. Pediatrics 54: , Gaskill SJ, Marlin AE: Pseudocysts of the abdomen associated with ventriculoperitoneal shunts: a report of twelve cases and a review of the literature. Pediatr Neurosci 15:23-26; discussion 6-7, Rainov N, Schobess A, Heidecke V, Burkert W: Abdominal CSF pseudocysts in patients with ventriculo-peritoneal shunts. Report of fourteen cases and review of the literature. Acta Neurochir (Wien) 127:73-78, Aparici-Robles F, Molina-Fabrega R: Abdominal cerebrospinal fluid pseudocyst: a complication of ventriculoperitoneal shunts in adults. J Med Imaging Radiat Oncol 52:40-43, Pernas JC, Catala J: Case 72: Pseudocyst around ventriculoperitoneal shunt. Radiology 232: , Salomo JF, Leibinger RD: Abdominal pseudocysts complicating CSF shunting in infants and children. Report of 18 cases. Pediatric Neurosurgery 31: , de Oliveira RS, Barbosa A, Vicente YA, Machado HR: An alternative approach for management of abdominal cerebrospinal fluid pseudocysts in children. Childs Nerv Syst 23:85-90, Nfonsam V, Chand B, Rosenblatt S, Turner R, Luciano M: Laparoscopic management of distal ventriculoperitoneal shunt complications. Surg Endosc 22 : , Esposito C, Colella G, Settimi A, Centonze A, Signorelli F, Ascione G, Palmieri A, Gangemi M: One-trocar laparoscopy: a valid procedure to treat abdominal complications in children with peritoneal shunt for hydrocephalus. Surg Endosc 17: , 2003

7 202 소아외과제 16 권제 2 호 2010 년 Abdominal Cerebrospinal Pseudocyst: a Complication of Ventriculoperitoneal Shunt in a Child Yoon-Jung Boo, M.D. Division of Pediatric Surgery, Department of Surgery, Korea University College of Medicine, Seoul, Korea Abdominal cerebrospinal fluid pseudocyst is an uncommon complication of ventriculoperitoneal shunt (VPS) performed for hydrocephalus. The incidence of VPS complications in children is higher than in adults. There are controversies and difficulties in the treatment of the abdominal pseudocyst. We report a case of abdominal pseudocyst complicating VPS in a boy. Partial excision of pseudocyst and replacement of the VP shunt were effective during a followup of 18 months postoperatively with no recurrence. (J Kor Assoc Pediatr Surg 16(2):196~202), Index Words:Ventriculoperitoneal shunt, Pseudocyst, Complication Correspondence:Yoon-Jung Boo, M.D., Division of Pediatric Surgery, Department of Surgery, Korea University College of Medicine 126-1, Anam-dong, Sungbuk-gu, Seoul , Korea Tel : 02) , Fax : 02) drboo@korea.ac.kr

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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,