Small duct primary sclerosing cholangitis (PSC) is characterized by cholestatic liver function tests, histological evidence of PSC but absence of classic cholangiographic findings. Large duct or classic PSC in association with hepatitis C virus (HCV) infection has rarely been reported. However to the best of our knowledge small duct PSC in association with HCV infection has not been reported. We report this case of small duct PSC in a patient with HCV infection. HCV infection in our patient was successfully treated with ribavirin and peg interferon alfa-2a, as evidenced by undetectable HCV ribonucleic acid levels. However, the patient had persistently elevated liver function tests suggestive of cholestasis. Endoscopic retrograde cholangiopancreatography (ERCP) revealed normal architecture of bile ducts. Hence patient underwent liver biopsy and its histopahological findings were suggestive of PSC. He had colonoscopy along with biopsy and inflammatory bowel disease (IBD) was ruled out.