CLINICAL RESEARCH e-ISSN 1643-3750 © Med Sci Monit, 2017; 23: 1371-1377 DOI: 10.12659/MSM.900313

Reoperation After Cyst Excision with Hepaticojejunostomy for Choledochal Cysts: Our Experience in 18 Cases

Received: 2016.06.28 Accepted: 2016.08.16 Published: 2017.03.20

Authors’ Contribution: Study Design  A Data Collection  B Analysis  C Statistical Data Interpretation  D Manuscript Preparation  E Literature Search  F Funds Collection  G

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Qingfeng Sheng Zhibao Lv Weijue Xu Xianmin Xiao Jiangbin Liu Yibo Wu

1 Department of General Surgery, Shanghai Children’s Hospital, Shanghai Jiao Tong University, Shanghai, P.R. China 2 Department of Surgery, Children’s Hospital, Fudan University, Shanghai, P.R. China

Zhibao Lv, e-mail: [email protected], [email protected] This study was supported by grants from the Science and Technology Commission of Shanghai Municipality (15ZR1434200)

Complete cyst excision with biliary reconstruction is the treatment of choice for choledochal cyst (CC). The aim of this article is to review our experience in patients who underwent reoperation between January 1995 and December 2014. The records of 18 patients (female/male, 15/3) were retrospectively analyzed including age, sex, cyst type, initial procedure, lab and imaging findings, indications for reoperation, intraoperative findings, and results of reoperation. The median follow-up period was 6 years. The rate of reoperation in this study was about 6.8%. Eighteen patients (7 type Ia, 2Ic, 9 IV-A) developed severe postoperative complications and required surgical intervention. The median time interval from primary surgery to reoperation was 19.5 months (range, 3 days to 8 years). Two early complications required surgery due to anastomotic bile leakage and intussusception. Sixteen late complications occurred, including 3 intrahepatic bile duct stenosis with calculi, 5 anastomotic strictures with/without stones, 4 intrapancreatic cyst remnants, 3 adhesive bowel obstructions, and 1 internal hernia. For patients with persistent dilatation of the intrahepatic bile duct or anastomotic stricture, removal of stones and revision of hepaticojejunostomy were performed, with additional hepatic ductoplasty when necessary. Radical excision of the dilated cystic remnant in the head of pancreas was performed in 4 patients, with 1 needing additional pancreaticojejunostomy procedure. No deaths occurred. Sixteen patients recovered uneventfully after reoperation, and 2 wound infections developed. A wide hepaticojejunostomy with/without ductoplasty is essential to prevent cholangitis, anastomotic stricture, and calculi formation. Complete cyst excision, including the intrapancreatic portion, should be meticulously pursued. Choledochal Cyst • Postoperative Complications • Reoperation http://www.medscimonit.com/abstract/index/idArt/900313

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Sheng Q. et al.: Reoperation for choledochal cyst © Med Sci Monit, 2017; 23: 1371-1377

CLINICAL RESEARCH

Background Choledochal cyst (CC) is a congenital dilatation of the extrahepatic and/or intrahepatic bile duct, and is commonly associated with pancreaticobiliary maljunction. Cyst excision combined with hepaticoenterostomy (hepaticoduodenostomy or hepaticojejunostomy) has become the standard procedure for children with choledochal cyst [1–3]. The majority (over 85–90%) have satisfactory surgical outcomes. However, 6–10% of patients need reoperative surgery due to severe postoperative complications, including bile leakage, anastomotic stricture, intrahepatic cholelithiasis, recurrent pancreatitis, adhesive bowel obstruction, and malignancy [2,4,5]. The aim of this study was to review our experience with 18 patients who underwent reoperation over the past 20 years.

Material and Methods Between January 1995 and December 2014, 275 children with CC were diagnosed and surgically treated in 2 tertiary hospitals (Children’s Hospital of Shanghai Jiao Tong University and Children’s Hospital of Fudan University). Cysts were categorized according to Todani’s classification: type Ia (n=176), type Ic (n=64), type III (n=1), type IV-A (n=33), and type V (n=1). The study was approved by the Institutional Review Board and the Ethics Board of Children’s Hospital of Shanghai Jiao Tong University and Children’s Hospital of Fudan University. Written informed consents were obtained from the parents. Regular follow-up was carried out by phone or interview in our clinics by an experienced surgeon (Y. Wu). In this study, procedures performed after primary surgery, such as percutaneous peritoneal drainage (n=2) and gastroscopy (n=1), were excluded. The medical records of 18 patients who underwent reoperation were retrospectively analyzed, including sex, cyst type, initial procedure, age at time of primary surgery and reoperation, clinical presentations, lab and imaging findings, indications for reoperation, intraoperative findings, and type and results of reoperation. Data were recorded as numeric or nominative variables. SPSS 17.0 software package (SPSS Inc, Chicago, IL) was used for statistical analysis. Differences in frequency distribution were compared using the c2 test or Fisher’s exact test. Other results are expressed as mean ± standard deviation (SD). A P-value of less than 0.05 was considered significant (a=0.05, two-tailed).

Results Initial operation was performed by open or laparoscopic cyst excision and end-to-side hepaticojejunostomy using a 30- to 40cm Roux limb without additional anti-reflux procedure. Enteric drainage (Roux-en-Y cystojejunostomy) was abandoned after

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January 1995. Twelve children (8 type Ia, 4 type Ic) were lost to follow-up. Of the remaining 263 patients, 43 children developed early (less than 30 days, n=18) or late (greater than 30 days, n=25) postoperative complications. Eighteen (6.8%, 15 females) children required surgical intervention (Table 1). They underwent definitive primary surgery (16 open, 2 laparoscopic) at the median age of 6 years (range, 11 months to 11 years). Diagnostic workup before reoperation was carried out, including thorough history-taking, meticulous clinical examination, lab and radiological studies (abdominal X-ray film, ultrasonography, computed tomography (CT), and magnetic resonance cholangiopancreatography (MRCP). The median time interval from primary surgery to reoperation was 19.5 months (range, 3 days to 8 years). Abdominal pain, fever, jaundice, and vomiting were seen in the great majority of cases. There was no significant difference in reoperation rate between open and laparoscopic procedure (7.5%, 16/212; 3.9%, 2/51; P=0.539). Two early postoperative complications required surgery due to anastomotic bile leakage (1 case) and intussusception (1 case) (Table 2). A 38-month-old girl presented with fever and abdominal pain 6 days after the primary procedure. A CT scan showed perihepatic fluid collection and eventration of the diaphragm on the right side (Figure 1). Ultrasonography-assisted paracentesis demonstrated bilious ascites. The patient recovered after leakage repair and peritoneal drainage. In the other patient, an 11-month-old girl, vomiting developed 3 days after primary surgery and intussusception was diagnosed by ultrasonography and CT scan (Figure 2). Laparotomy revealed ileo-ileal intussusception and manual reduction was performed. Other early complications were managed conservatively with good results, including wound infection (n=4), wound dehiscence (n=1), gastrointestinal bleeding (n=2), chylous ascites (n=3), acute pancreatitis with pancreatic juice leakage (n=1), anastomotic leakage (n=2), and intra-abdominal infection or abscess formation (n=3). Twenty-five late complications occurred and 16 of these patients underwent relaparotomy (Table 3), including intrahepatic bile duct stenosis with stones (n=3), anastomotic stricture with or without stone formation (n=5), intrapancreatic cyst remnant (n=4), adhesive bowel obstruction (n=3), and internal hernia formation (n=1). No malignant change was observed. Postoperative cholangeitis occurred significantly more often in type IV-A than in type I patients (27.2%, 9/33; 1.3%, 3/228; P

Reoperation After Cyst Excision with Hepaticojejunostomy for Choledochal Cysts: Our Experience in 18 Cases.

BACKGROUND Complete cyst excision with biliary reconstruction is the treatment of choice for choledochal cyst (CC). The aim of this article is to revi...
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