The patient was a 44 year-old female who was given an oral contraceptive (Anovlar) because of myoma of uterus. After taking approximately 50 tablets, itching of skin and jaundice appeared. Jaundice persisted more than half a year and rxanthoma, positive antimitochondrial antibody and elevated IgM were noted. Liver biopsy revealed periportal fibrosis, cholestasis and atypical proliferation of bile ductules, but failed to show chronic nonsuppurative destructive cholangitis (CNSDC). Electron microscopy of the biopsied liver specimens showed diminished microvilli and bleb formation in the dilated bile canaliculi. Cholesterol cyrstals were also noted in the cytoplasm. Although lymphocyte blast formation test by anovlar was negative, this case suggests the close relation between drug-induced cholestasis and primary biliary cirrhosis.