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Cholestatic jaundice caused by imidazole derivatives is a rare complication of antithyroid drug therapy. We present a case of cholestatic jaundice with systemic hypersensitivity reaction, which developed in a 27-year old male one day after exposure to methimazole. The patient showed clinical improvement and gradual resolution of jaundice after the discontinuation of methimazole and treatment with prednisolone. Histologic findings of liver revealed bile pigment, predominantly in centrilobular area, and infiltration of chronic inflammatory cells in a few portal area without evidence of degeneration or necrosis of hepatocytes. Methimazole could be presumed as etiologic agent from clear chronological relationship and the lack of other causative factors. We report this unusual case with review of literature.