Primary Sclerosing Cholangitis (PSC) is a rare cholestatic condition seen locally. Its etiology is unknown and it is commonly associated with ulcerative colitis, another rare condition seen locally. In this report, 2 patients with PSC, both Indian males, had ulcerative colitis and in one PSC was diagnosed some 15 years later. An interesting feature common to both patients was that of intra-hepatic ductal involvement. Percutaneous transhepatic cholangiogram (PTC) and Endoscopic Retrograde Cholangio-Pancreatogram (ERCP) showed characteristic stricturing and beading of the intra-hepatic ducts.