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1 pissn eissn Joo Y, et al: Laparoscopic Nissen Fundoplication in Neurologically Impaired Children vs. Neurologically Normal J Korean Assoc Pediatr Surg Vol. 22, No. 2, December Original Article 신경학적손상여부에따른소아에서의복강경하 Nissen 위바닥주름술 주요한 1, 이주연 1,2, 최정현 1,2, 남궁정만 1,2, 김성철 1,2, 김대연 1,2 1 울산대학교의과대학외과학교실, 2 서울아산병원어린이병원소아외과 Laparoscopic Nissen Fundoplication in Children with Neurological Impairment versus Neurologically Normal Children Yohan Joo 1,2, Ju Yeon Lee 1,2, Jung Hyun Choi 1,2, Jung-Man Namgung 1,2, Seong-Chul Kim 1,2, Dae Yeon Kim 1,2 1 Department of Surgery, University of Ulsan College of Medicine, 2 Department of Pediatric Surgery, Asan Medical Center Children s Hospital, Seoul, Korea Purpose: This study aimed to assess the long-term clinical outcome of laparoscopic Nissen fundoplication (LNF) in children according to their neurologic status. Methods: The study retrospectively analyzed the data of 82 children (62 neurologically impaired and 20 neurologically normal children with primary gastroesophageal reflux disease) who had undergone LNF between 2003 and The main outcome measures were the occurrence of recurrence that required reoperation and post-procedure complications such as infections, pneumonia, and gastrointestinal complications including ileus, dysphagia, and delayed gastric emptying. Results: The median age at the time of the LNF was 25 months (range, months), and the median of body weight was 10.0 kg (range, kg). The average weight gain was 1.55±1.68 kg at 6 months, 3.32±2.30 kg at 1 year, and 5.63±4.22 kg at 2 years after surgery. Six (9.7%) of the 62 neurologically impaired patients and two (10.0%) of neurologically normal lost their body weight or had no weight changes. Eight (12.9%) of the 62 neurologically impaired children had required redo surgery because of gastroesophageal reflux disease recurrences, while 2 (10.0%) of the 20 neurologically normal children had experienced recurrences. In the neurologically impaired children, the postoperative complications included pneumonia (n=1), wound infection (n=1), urinary tract infection (n=1), dysphagia (n=1), delayed gastric emptying (n=1), and ileus (n=2). All of these complications were not found in the neurologically normal group, except for only one case of infectious colitis. However, there was no statistically significant difference between the two groups in postoperative complications. Conclusion: The outcomes of laparoscopic fundoplication were similar in the neurologically impaired children and in the neurologically normal children. Keywords: Gastroesophageal reflux, Laparoscopy, Fundoplication, Neurologic deficit 서론소아에서의복강경하위바닥주름술은위식도역류에대하여최근가장선호되는수술적치료이다 [1]. 일반적으로신경학적손상이있는환자들은정상인환자들에비해위식도역류방지를위한위바닥주름술후합병증이더빈번하게발생하는것으로알려져있다 [2,3]. Pearl 등 [4] 은신경학적손상이있는환자들이손상이없는환자들에비해위바닥주 름술후재수술을시행한비율이 4배더높다고보고하기도하였다. 하지만이러한보고들은개복을통해시행한위바닥주름술에따른결과들을비교한연구들이며신경학적손상이위바닥주름술후합병증을증가시키는원인에대하여는명확하게제시하지못했다. 2003년 Esposito 등 [5] 의연구에서는신경학적손상을가진소아에서높은사망률을보였지만, 대부분의경우항역류수술과는관련이없었다고고찰한바있다. 최근들어위바닥주름술에도최소침습수술이도입 Received: August 19, 2016, Revised: October 22, 2016, Accepted: October 24, 2016 Correspondence: Dae Yeon Kim, Department of Pediatric Surgery, Asan Medical Center Children s Hospital, 88 Olympic-ro 43-gil, Songpa-gu, Seoul 05505, Korea. Tel: , Fax: , kimdy@amc.seoul.kr Copyright 2016 Korean Association of Pediatric Surgeons. All right reserved. This is an Open Access article distributed under the terms of the Creative Commons Attribution Non-Commercial License ( licenses/by-nc/4.0) which permits unrestricted non-commercial use, distribution, and reproduction in any medium, provided the original work is properly cited. Journal of Korean Association of Pediatric Surgeons 49

2 J Korean Assoc Pediatr Surg 2016;22(2):49-53 되었고발전되고있는단계로, 저자들도이미소아에서의복강경하위바닥주름술을통한위식도역류의수술적치료의결과를보고하며신경학적으로정상인환자중에서는재발이없었던반면, 신경학적손상을가진환자중 2명이재발로재수술을받아상대적으로높은재수술률을확인한바있다 [6]. 이러한보고후 10년이지난시점에서, 복강경하위바닥주름술이시행된위식도역류소아환자에서신경학적손상을동반한환자가손상을동반하지않은환자에비해수술후재발로인한재수술및합병증이더많은지를알아보고자하였다. 대상및방법울산대학교의과대학서울아산병원에서 2003년 1월부터 2012년 12월까지 10 년간최소침습수술로복강경하위바닥주름술을받은 18세이하의소아환자에대하여의무기록을바탕으로후향적연구를시행하였다. 위식도역류의진단은지속적인구토나반복적인폐렴을보이는환자에서환자의전신상태에따라상부소화관내시경, 24 시간식도산도검사, 비디오연하조영검사, 식도조영술을선택적으로시행하여확인하였다. 수술후합병증중감염성합병증및폐렴은수술과관련없이발생한경우를구분하기위해수술후 3개월이내에진단된경우로기간을한정하였다. 모든수술은전신마취하에복강경하 Nissen 위바닥주름술이시행되었다. 첫번째투관침은배꼽상방에환상형의피부절개후삽입하였다. 첫번째투관침을삽입한후환자의체격을고려하여 3 mm 나 5 mm 투관침에 30도카메라를넣고전체복강을확인하였다. 이후우상복부에간의견인과기구조작을위해두개의투관침, 좌상복부에도수술기구조작을위해두개의투관침을넣어총 5개의투관침을사용하여수술을시행하였다. 이후저자들이시행한 Nissen 위바닥주름술은저자들의기존의보고와같다 [6]. 상복부의투관침을통해간좌엽을우측으로견인하여횡격막및위-식도경계를노출시켜식도열공탈장 (esophageal hiatal hernia) 여부를확인하였다. 식도열공탈장이동반된경우에는탈장교정술을같이시행하였으나, 첨포 (patch) 가필요한경우는없었다. 미주신경을확인하여보존하며횡격막각 (diaphragmatic crus) 을박리하여복부식도의길이를최소 2 cm 이상확보하였다. 횡격막각의근육을 Ethibond (Ethicon, Pittsburgh, PA, USA) 를사용하여봉합하여고정하였다. 유동화된위바닥후면부위를식도의뒤쪽창을통해시계방향으로돌려식도를감싼뒤 2회내지 3 회정도봉합하였다. 위루관이필요한경우에는좌상복부의 투관침을통하여삽입하였다. 대상기간동안복강경하위바닥주름술이계획되었던환자는 89명이었으나, 이중 7명은수술중개복술로전환되어이를제외한 82명을대상으로연구하였다. 이중신경학적손상이있었던환자는 62명이었으며, 그원인은 Table 1과같다. 기초통계는 t검정을사용하여비교하였으며, 수술후합병증발생률은 χ 2 검정으로분석하였다. 모든통계검정에서 p값이 0.05 미만인경우를통계적으로유의한것으로보았다. 모든분석은 IBM SPSS Statistics 20.0 (IBM Co., Armonk, NY, USA) 을사용하여수행하였다. 결과 전체 82명중남자는 50명, 여자는 32명이었다. 신경학적손상을동반했던환자 ( 손상군 ) 62명중남자는 40명, 여자는 22명이었으며, 신경학적손상이없었던환자 ( 정상군 ) 20명 Table 1. Causes of Neurologic Impairment Neurologic impairment No. of patients Acquired neurologic impairment Perinatal asphyxia 2 Hypoxic brain damage 3 Encephalitis 4 Traumatic ICH 2 Medulloblastoma 2 Brain stem glioma 1 Congenital neurologic impairment Anencephaly 1 Microcephaly 1 Leukodystrophy 1 Degenerative neuromuscular disease 3 Congenital rubella 1 Agenesis of corpus callosum 1 Syndrome 18q22 deletion 1 Down syndrome with perinatal asphyxia 1 Werdnig-Hoffman 1 Seizure disorder 13 CHARGE syndrome 3 3P deletion 1 Lennox-Gastaut syndrome 2 Noonan syndrome 1 Adreno-leuko-dystrophy 1 Krabbe disease 1 Cerebral palsy 9 Costello syndrome 1 Leigh syndrome 4 Pelizaeus-Merzbacher disease 1 Total 62 ICH, Intracranial hemorrhage. 50 Journal of Korean Association of Pediatric Surgeons

3 Joo Y, et al: Laparoscopic Nissen Fundoplication in Neurologically Impaired Children vs. Neurologically Normal 중남자가 10명, 여자가 10명이었다. 전체환자의수술시나이는중간값 25개월 ( 범위 1-192개월 ) 이었고, 신경학적손상군의나이는중간값 30개월 ( 범위 개월 ), 신경학적정상군은중간값 10.5 개월 ( 범위 개월 ) 이었다. 수술시체중은전체환자에서중간값 10.0 kg ( 범위 kg) 이었으며, 신경학적손상군의경우중간값 11.1 kg ( 범위 kg), 신경학적정상군은중간값 9.0 kg ( 범위 kg) 이었다 (Table 2). 수술시간은평균 145.8±50.1 분이었으며, 신경학적손상군과정상군은각각 148.1±54.0 분과 138.7±39.7 분으로신경학적손상군이더길었지만통계적으로유의한차이는없었다 (p=0.319). 수술후연식식이까지의기간은신경학적손상군은평균 4.0±2.2 일 ( 범위 1-12 일 ), 중간값 4일이었고, 정상군에서는평균 3.3±2.0 일 ( 범위 1-10 일 ), 중간값 3일이었다. 단순비교로는약 1일정도의차이를보이고있지만, 통계적으로의미는없었다 (p=0.857). 체중변화를확인한결과, 전체환자에서수술후 6개월뒤평균 1.55±1.68 kg, 1년뒤평균 3.32±2.30 kg, 2년뒤평균 5.63±4.22 kg의체중증가를보였다. 각군별로는신경학적손상군에서수술후 6개월뒤평균 1.68±1.76 kg, 1년뒤 3.23±2.07 kg, 2년뒤 5.05±3.28 kg의체중증가를보였으며, 신경학적정상군에 서는수술후 6개월뒤 1.14±1.36 kg, 1년뒤 3.68±3.07 kg, 2년뒤 7.56±6.26 kg의체중증가가확인되었다. 6개월및 1년경과시점에서는두군간체중증가의유의한차이가없었으나, 2년경과시점에서는신경학적정상군이신경학적비정상군에비해유의하게더많은체중증가를보였다 (p=0.024). 수술 1년경과시점에서체중감소를보였거나, 체중변화가 1 kg 이하로변화가없었던환자는전체 8명 (9.8%) 으로신경학적손상군에서 6명 (9.7%), 신경학적정상군에서 2명 (10.0%) 으로유의한차이가없었다 (p=0.629; Table 3). 수술후감염성합병증은신경학적손상군에서는창상감염, 요로감염이각각 1명씩발생되었다. 신경학적정상군에서는 1명의환자가감염성장염으로수술후 2주뒤입원치료를필요로했던반면, 창상감염, 요로감염은한명도발생되지않았다. 호흡기계합병증은신경학적손상군에서 1명의환자가수술후폐렴으로치료를받은반면, 신경학적정상군에서는수술후 3개월내에폐렴이진단된환자는없었다. 위장관계합병증은신경학적손상군에서수술후연하곤란을호소한환자가 1명, 지연위배출로식이진행이늦어진환자가 1명씩있었고, 장폐색으로수술이필요한환자가 2명발생되었다. 신경학적정상군에서는한명도발생되지않았다. 위식도역류증이재발되어재수술이필요했던경우는총 10 Table 2. Clinical Characteristics of Patients Neurologically normal Neurological impaired All patients p-value Patient Sex (male:female) 10:10 40:22 50: Age at operation (mo) 10.5 (2-137) 30 (1-192) 25 (1-192) Body weight at operation (kg) 9.0 ( ) 11.1 ( ) 10.0 ( ) Values are presented as n only or median (range). Table 3. Results of Operation Neurologically normal (n=20) Neurological impaired (n=62) All patients (n=82) p-value Operation time (min) 138.7± ± ± Gastrostomy 13 (65.0) 56 (90.3) 69 (84.1) <0.05 Time to diet (day) Mean 3.3± ± ± Median (range) 3 (1-10) 4 (1-12) 3 (1-12) Body weight gain (kg) 6 mo 1.14± ± ± yr 3.68± ± ± yr 7.56± ± ± Weight loss or no change at 1 year after operation 2 (10.0) 6 (9.7) 8 (9.8) Values are presented as mean±sd, n (%), or median (range). Journal of Korean Association of Pediatric Surgeons 51

4 J Korean Assoc Pediatr Surg 2016;22(2):49-53 Table 4. Postoperative Complications Neurologically normal (n=20) Neurologic impaired (n=62) All patients (n=82) p-value Infection 1 (5.0) 2 (3.2) 3 (3.7) Wound infection 0 (0) 1 (1.6) 1 (1.2) Infectious colitis 1 (5.0) 0 (0) 1 (1.2) Urinary tract infection 0 (0) 1 (1.6) 1 (1.2) Respiratory complication Pneumonia 0 (0) 1 (1.6) 1 (1.2) Gastrointestinal complication 0 (0) 4 (6.5) 4 (4.9) Dysphagia 0 (0) 1 (1.6) 1 (1.2) Delayed gastric emptying 0 (0) 1 (1.6) 1 (1.2) Ileus 0 (0) 2 (3.2) 2 (2.4) Recurred gastroesophageal reflux 2 (10.0) 8 (12.9) 10 (12.2) Values are presented as n (%). 명 (12.2%) 으로, 이중신경학적손상군에서 8명 (12.9%), 정상군에서는 2명 (10.0%) 에서발생되었으나두군간통계적으로유의한차이는없었다 (p=0.730). 재수술이시행된모든환자에서위바닥주름술부위의느슨해짐이관찰되었다. 수술후사망예는총 10명으로, 신경학적손상군에서 9명, 정상군에서 1명이있었으나모두수술과는관련없는기저질환으로인한사망이었으며, 수술시점으로부터 1년이상경과한이후에사망하였다. 신경학적손상군중 5명은호흡부전으로, 1명은심부전, 1명은수모세포종 (medulloblastoma) 의악화, 1명은두개내출혈로사망하였으며, 1명은타원에서치료중사망하여사망의원인을알수없었다. 신경학적정상군에서사망한 1명은심방중격결손증, 동맥관개존증을가진환자로심부전에동반된폐동맥고혈압의악화로사망하였다 (Table 4). 고찰위식도역류를가진소아에서의복강경하 Nissen 위바닥주름술을시행한결과신경학적손상여부와관계없이대체로체중이증가되었다. 일부환자에서는체중이증가하지않거나오히려감소되기도하였는데, 이는기저질환의악화나심각한발육장애가동반된경우였으며, 신경학적손상여부와관계없이비슷한비율로관찰되었다. 수술후합병증에대해서는기존연구에서신경학적손상을가진환자에서수술후합병증이더많다는보고가대부분이었지만, 본연구에서는두군간에유의한차이는없었다. 이는크게두가지요인으로생각해볼수있는데, 첫째로수술기법의발전및전반적인의료의질적발달로이전에비해신경학적손상을가진환자에서의결과가향상된것으로고려된다. 둘째로, 본연구가이루어진기관은상급종합병원으 로신경학적손상이없는환자라하더라도메틸말론산혈증 (methylmalonic acidemia) 으로간이식을받았던환자이거나, 선천성심장기형을동반하거나, 기타희귀중증질환을가진환자들이포함되어있어, 표본선택에있어편향이작용되었을가능성을배제할수없다. 수술후식이진행까지의소요기간도두군간에유의미한차이가없어, 신경학적손상의동반여부가수술후식이까지의걸리는시간에미치는영향은중요하지않은요소로분석되었다. 두군모두에서수술후 1일만에연식식이가가능한환자가있었던반면, 위의지연위배출이나감염등수술후환자상태에따라 10일이상소요되는경우도있었다. 수술후위식도역류증이재발되어추가적인재수술이필요한경우도두군간차이는없었으며, 전체환자에서의재수술이필요했던경우는 12.2% 로이전의다른연구들과비슷한정도의재발률을확인할수있었다 년 Diaz 등 [7] 의연구에따르면복강경하 Nissen 위바닥주름술후 2년내에재수술이필요했던경우는 13.4% 로보고하고있다. 항역류수술후위식도역류의재발은수술자의기술적인숙련도와식도열공탈장동반여부등과관련이깊은것으로알려져있다. 본연구에서위식도역류를겪고있는소아를대상으로시행된복강경하 Nissen 위바닥주름술의합병증및재발률은이전의연구와비슷한결과를보였으며, 신경학적손상을동반한환자와동반하지않았던환자사이의유의한차이는없었다. 신경학적손상의동반여부가수술후결과에주는영향은미미한것으로보인다. 이는기존의타연구에서도일관되게보여지고있는결과이다 [8]. 그러나환자를신경학적손상군으로분류할때여러종류, 다양한중증도의질환이포함되게되는반면, 심각한기저질환으로인해거동불가상태이면서도신경학적정상아로분류되는경우도있었다. 이런점을고려해볼때단순히신경학적손상여부만으로분류하 52 Journal of Korean Association of Pediatric Surgeons

5 Joo Y, et al: Laparoscopic Nissen Fundoplication in Neurologically Impaired Children vs. Neurologically Normal 기보다는환자의전신상태, 특히거동가능여부나동반된기저질환의중증도를기준으로평가하는것이더바람직할것으로생각되며, 임상에서는각환자의상태에대해개별적으로접근하여치료에적용해야할것이다. 아직까지국내에서소아에서의위식도역류의수술적치료에대한연구는경험에대한후향적보고에치중되어있다 [9]. 이제국내에서도어느정도경험이축적된만큼앞으로는지금까지의경험을바탕으로전향적연구를시도해볼수있을것으로기대된다. 결론적으로, 본연구에서는신경학적손상군에서시행한복강경 Nissen 위바닥주름술후합병증및회복경과, 수술후재발률은신경학적정상군과별다른차이가없었다. CONFLICTS OF INTEREST No potential conflict of interest relevant to this article was reported. REFERENCES 1. Mattioli G, Esposito C, Lima M, Garzi A, Montinaro L, Cobellis G, et al. Italian multicenter survey on laparoscopic treatment of gastro-esophageal reflux disease in children. Surg Endosc 2002;16: Martinez DA, Ginn-Pease ME, Caniano DA. Sequelae of antireflux surgery in profoundly disabled children. J Pediatr Surg 1992;27: ; discussion Flake AW, Shopene C, Ziegler MM. Anti-reflux gastrointestinal surgery in the neurologically handicapped child. Pediatr Surg Int 1991;6: Pearl RH, Robie DK, Ein SH, Shandling B, Wesson DE, Superina R, et al. Complications of gastroesophageal antireflux surgery in neurologically impaired versus neurologically normal children. J Pediatr Surg 1990;25: Esposito C, Van Der Zee DC, Settimi A, Doldo P, Staiano A, Bax NM. Risks and benefits of surgical management of gastroesophageal reflux in neurologically impaired children. Surg Endosc 2003; 17: Nam SH, Kim DY, Kim SC, Kim IK. Laparoscopic nissen fundoplication in children for treatment of gastroesophageal reflux disease. J Korean Assoc Pediatr Surg 2007;13: Diaz DM, Gibbons TE, Heiss K, Wulkan ML, Ricketts RR, Gold BD. Antireflux surgery outcomes in pediatric gastroesophageal reflux disease. Am J Gastroenterol 2005;100: Capito C, Leclair MD, Piloquet H, Plattner V, Heloury Y, Podevin G. Long-term outcome of laparoscopic Nissen-Rossetti fundoplication for neurologically impaired and normal children. Surg Endosc 2008;22: Kim ST, Lee CK, Kim HE, Seo JM, Lee SK. The eleven years experience with fundoplication in infants and children. J Korean Assoc Pediatr Surg 2008;14: Journal of Korean Association of Pediatric Surgeons 53

황지웅

황지웅 Comparison of Laparoscopy and Exploration in the Distal Pancreatectomy BACKGROUND: To determine the benefits of laparoscopic surgery compared with exploration, the clinical outcomes of open and laparoscopic