Carbimazole is a drug commonly used to treat thyrotoxicosis. Among its adverse effects, cholestatic jaundice is uncommon. There is no definitely particular risk factors, and its occurrence could not be predicted before treatment. This complication is supposed to be an immune reaction. Here, we report a 54 year-old female without history of hepatitis before, was admitted due to skin itching and jaundice 5 weeks after carbimazole (20mg/day) used for treatment of thyrotoxicosis. After switching to propylthiouracil (PTU) (200 mg/day), the thyroxine level returned to normal, but the jaundice became more apparent, so PTU was also withdrawn. The thyroxine level elevated again 4 weeks later, she was then ablatively treated with radioactive iodine and propranolol. The total bilirubin level returned to normal 28 weeks after withdrawal of carbimazole. She became to be hypothyroid status 78 weeks after the first visiting. The pathogenesis of carbimazole-induced cholestatic jaundice is still unclear, and there is no effective medical therapy. Early recognition and withdrawal of anti-thyroid medication is pivotally important.