Background Primary intrahepatic bile duct dilatation can be very harmful to patients although it belongs to benign biliary disease. It can occur in any part of the liver, intraoperative laparoscopic ultrasound (LUS) guidance combine with real-time indocyanine green (ICG) fluorescence navigation are the means of choice for accurate surgical resection. Case presentation Herein we reported a 43-year-old female patient presented with repeated right upper abdominal pain and distension for 3 years

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... aggravated for half a year, without fever and jaundice. A diagnosis of localized bile duct dilatation with lithiasis in segment 4 (S4) was made on the basis of preoperative imaging. Correspondingly, we selected to perform a laparoscopic surgery with LUS guided real time ICG fluorescence imaging (ICG-FI) and navigation to make the operation more simply and accurately, as well as to retain normal tissues in a certain extent. Laparoscopic resection of S4b and partial S4a was successfully performed, without any complications. Conclusion Laparoscopic anatomical surgery for intrahepatic bile duct dilatation is a technically challenging operation. The combined use of preoperative three-dimensional computerized tomography (CT) planning, intraoperative LUS guided super-selection, ICG hepatic segment staining and real-time fluorescence navigation could help surgeons accurately complete the segmentectomy or subsegmentectomy with minimized trauma and maximized liver tissue preservation.