Introduction: Laparoscopic cholecystectomy (LC) is the gold standard treatment of cholelithiasis. However, LC is associated with slightly higher rate of complications than laparotomy. To perform a safe LC, it is important to exercise caution for an abnormal course of hepatic artery though the incidence itself is low. Herein, we report a rare case of LC in which medial segment artery mimicking cystic artery. To the best of our knowledge, such a case has not been reported. Case presentation: A 35-year-old male visited our hospital with a complaint of epigastric pain. The symptom had continued for two months. Ultrasound and computed tomography revealed cholelithiasis. Magnetic resonance image did not show any biliary abnormalities. The patient was scheduled for an elective laparoscopic cholecystectomy. At the time of surgery, the gallbladder was slightly inflamed. After dissection of the Calot's triangle, the cystic duct and a single large artery were identified. Though the critical view of safety was confirmed at first glance, dissection of periarterial adhesions revealed that the artery ran into the medial segment of the liver. Further dissection revealed short double cystic arteries branched from the aberrant artery. After the confirmation of all of the abnormal course of the arteries, the operation has been performed safely without arterial injury. The patient made a satisfactory recovery, and was discharged on 3 days after operation. Conclusion: The knowledge of the anatomy of the cystic artery and careful dissection are an important aspect in achieving a complication free LC.

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