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Methimazole is a widely used and generally well-tolerated antithyroid agent. Adverse reactions occur in 1~5% of patients taking methimazole medication, but these are most commonly transient, benign leukopenia and a skin rash. Severe cholestatic jaundice, combined with agranulocytosis, has been known as a rare complication. Herein, a case of methimazole induced cholestatic jaundice, with agranulocytosis, is reported.