Recurrence of primary sclerosing cholangitis (PSC) after liver transplantation is very uncommon. The true incidence of recurrence is unknown, mainly because of the difficulty in differentiating ischemic strictures from that of recurrent disease. Primary sclerosing cholangitis and ischemic strictures have identical histopathologic and cholangiographic features. We report a young man who had recurrence of PSC in two allografts and report our experience in 32 patients who had liver transplantation for PSC. Six patients (18%) had evidence of non-anastomotic strictures and, of these, only one patient (reported here) had unequivocal evidence of true recurrence. The strictures in other five patients happened because of ischemia. The recurrence of the disease in two allografts in an immunosuppressed patient, in the absence of ischemia, chronic rejection, or any known pathogen, raises the question of the role of an unidentified infectious agent in the etiopathogenesis of PSC.