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Background: Traumatic biliary neuromas (TBNs) represent a rare cause of biliary stricture (BS) after orthotopic liver transplantation (OLT). Diagnosis is challenging preoperatively and is most often made at pathology after resection. Herein, we report a 20-year experience of TBN-related BS., Patients and Methods: Medical records of 1030 adult patients undergoing OLT from 1991 to 2014 were reviewed. Patients with histologically proven TBN were identified among those presenting a BS., Results: Over the study period, 52 patients developed an anastomotic BS. Of these, 17 had repeat surgery and specimen examination identified TBN in five instances. All five patients with TBN had a duct-to-duct biliary reconstruction during OLT. Median delay from OLT to onset of symptoms was 69 months (range 4-239). Preoperative imaging showed a compressive mass in one patient. Four patients underwent TBN resection combined with hepaticojejunostomy and had an uneventful postoperative course. One patient underwent TBN resection and duct-to-duct reconstruction; he died from acute pancreatitis on postoperative day 21. After a median follow-up of 40.5 months (range 10-54), no recurrent BS occurred., Conclusion: Traumatic biliary neuromas represent a possible diagnosis for unexplained anastomotic BS after OLT. Surgical excision combined with hepaticojejunostomy is effective, allows histological diagnosis, and prevents from recurrence., (© 2016 John Wiley & Sons A/S. Published by John Wiley & Sons Ltd.)