Abstract: Objective:To investigate the epidemiological characteristics, clinical features, diagnosis, treatment and prognosis of benign tumor of the bile ducts.
Methods:The retrospective crosssectional multicenter study was conducted. The clinical data of 136 patients with benign tumor of the bile ducts who were admitted to the eight hospitals between January 2007 and December 2016 were collected, including 70 in the Eastern Hepatobiliary Surgery Hospital of the Second Military Medical University, 19 in the First Affiliated Hospital of Zhengzhou University, 15 in the First Affiliated Hospital of Xi′an Jiaotong University, 11 in the First Affiliated Hospital of Dalian Medical University,7 in the Affiliated Hospital of North Sichuan Medical College, 6 in the Southwest Hospital of the Third Military Medical University, 4 in the Cancer Center of Sun YatSen University and 4 in the Xinhua Hospital Affiliated to Shanghai Jiao Tong University School of Medicine. All the patients received laboratory and imaging examinations, and then underwent corresponding surgery when treatment planning was respectively determined by comprehensive hospitals according to clinical features and results of examinations. Surgical procedures were performed based on the results of intraoperative frozen section in rapid pathological diagnosis. Observation indicators: (1) epidemiological characteristics; (2) clinical features; (3) results of laboratory and imaging examinations; (4) treatment situations; (5) followup situations. Followup using outpatient examination and telephone interview was performed to detect the survival of patients up to March 2017. Measurement data with normal distribution were represented as ±s .
Results:(1) Epidemiological characteristics: Of 136 patients, the male to female ratio was 1.78∶1. The incidence of whole bile duct tumors was from high to low, including 52 patients with duodenal papilla adenoma, 32 with extrahepatic bile duct adenoma, 24 with intrahepatic biliary cystadenoma, 11 with intrahepatic papillary adenoma, 9 with intrahepatic bile duct epithelial tumor, 7 with epithelial tumor of duodenal papilla and 1 with neuroendocrine tumor of duodenal papilla. Among 136 patients, adenomas (including cystadenoma) was detected in 108 patients, papillomas in 11 patients, intraepithelial neoplasias in 16 patients, neuroendocrine tumor in 1 patient; intrahepatic bile duct benign tumors in 44 patients and extrahepatic bile duct (including duodenal papilla) benign tumors in 92 patients. (2) Clinical features: of 44 patients with intrahepatic bile duct benign tumors, 29 had abdominal pain, fever and abdominal masses, 4 had jaundice, 11 had no obvious clinical symptoms and were diagnosed by physical examination. Among 92 patients with extrahepatic bile duct benign tumors, 76 developed obstructive jaundice, 68 were accompanied by abdominal pain or colicky pain, 8 were combined with pancreatitis and 2 developed hemobilia, some patients were combined with multiple clinical symptoms. (3) Results of laboratory and imaging examinations: 82 patients received CA199 test, results of 22 patients were abnormal, with a level of (148±126)U/mL. Ninetyseven patients received carcinoembryonic antigen (CEA) test, with a level of test of (2.7±2.0)μg/L, and a level of CEA in 1 patient was slight abnormal, with a level of 11.2 μg/L. One hundred and thirtysix patients underwent preoperative ultrasound examinations, showing unqualified hepatic and bile duct space occupying lesions and bile duct dilatation. Ninetyfive patients underwent preoperative computed tomography (CT), 80 underwent preoperative magnetic resonance imaging (MRI) or magnetic resonanced cholangiopancreatography (MRCP), and 13 underwent preoperative endoscopic ultrasonography (EUS). Twentynine patients were considered for intrahepatic bile duct benign tumors; 76 were diagnosed with obstructive jaundice, with uncertain benign or malignant tumors; other patients had bile duct space occupying, considering bile duct tumor (including cancer). Twelve patients with bile duct obstruction underwent ERCP, showing obstruction site and morphology. (4) Treatment situations: among 136 patients, 65 underwent pancreaticoduodenectomy, 17 underwent right hemihepatectomy+cholecystectomy, 16 underwent cholecystectomy+hepatic left lateral lobectomy, 11 underwent left hemihepatectomy+cholecystectomy, 11 underwent duodenal papillary local excision+papilla reconstruction, 11 underwent RouxenY choledochojejunostomy anastomosis, 4 underwent cholecystectomy+extrahepatic bile duct local excision+endtoend bile duct anastomosis and 1 underwent endoscopic mucosal resection of duodenal papillary adenoma. Of 136 patients with postoperative complications, 25 were complicated with pancreatic leakage, 11 with bile leakage, 2 with postoperative hemorrhage and 1 with hepatic failure. Two patients with pancreatic leakage died of massive hemorrhage caused by abdominal infection, 1 died of hepatic failure and other patients were discharged from hospital after symptomatic treatment. (5) Followup situations: 47 of 136 patients were followed up for 3-123 months, with a followup rate of 34.6%. During followup, 2 patients undergoing duodenal papillary local excision + papilla reconstruction had canceration, and other patients had good survival.
Conclusions:There is a low clinical incidence of benign tumor of the bile ducts, which is more common in male than in female, and in adenomas (including cystadenoma) and papillomas. The preoperative imaging examinations or ERCP biopsy pathological examination can increase an accuracy of preoperative diagnosis. Benign tumors with high canceration rates need positive surgical treatment.