BACKGROUND: Prepancreatic postduodenal portal vein is extremely rare, with only 13 cases reported in the literature. CASE PRESENTATION: A 55-year-old white woman presented to our emergency department with abdominal pain. She underwent a computed tomography of her abdomen, which showed a portal vein coursing anterolaterally to her pancreas and posteriorly to the first portion of her duodenum, constituting a prepancreatic postduodenal portal vein. Imaging revealed choledocholithiasis, requiring endoscopic sphincterotomy, but due to a history of a gastric bypass procedure, she was lost to follow-up after being referred to an advanced endoscopist. This represents the 14th reported case of prepancreatic postduodenal portal vein. CONCLUSIONS: Awareness of this rare anomaly is paramount, and will help surgeons and interventional radiologists to avoid complications related to inadvertent injury to the portal vein, which could be life-threatening.