Traumatic biliary neuroma after orthotopic liver transplantation: a possible cause of 'unexplained' anastomotic biliary stricture

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.