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Cumulative experience in deceased donor liver transplantation for end-stage liver disease due to primary sclerosing cholangitis (PSC) suggests that liver transplantation is the treatment of choice with excellent results. Reports on the outcome of live donor liver transplantation (LDLT) for PSC, however, remain anecdotal.The clinical course and genetic disposition of nine patients who underwent LDLT for PSC were analyzed. The median follow-up period was 3.5 years.Cumulated 5-year patient and graft survival rates were 90% and 71%, respectively. Recurrent PSC was diagnosed in four patients. Ratios of freedom from recurrent PSC at 1, 3, and 5 years were 100%, 73%, and 49%, respectively. The mean time to recurrence was 3.3 years. Excluding the one case with a biologically unrelated donor, recurrent PSC was diagnosed in 50% (4/8). None of the patients presented with the human leukocyte antigen-B8DR3 haplotype, which is associated with a higher susceptibility for developing PSC among the Caucasian population. Overall patient survival of LDLT for PSC seems to equal that of deceased donor liver transplantation.PSC might recur earlier at a higher ratio after LDLT. Further study with protocol cholangiogram and genetic considerations, including high resolution human leukocyte antigen haplotype analysis, is necessary.