There has been little information regarding the pharmacokinetics of antithyroid drugs in patients with endstage renal disease (ESRD). We report here the pharmacokinetics and dialyzability of the antithyroid drug thiamazole in a chronic hemodialysis patient with hyperthyroidism. The patient was a 46-year-old woman who complained of palpitations 3 years after starting chronic hemodialysis therapy, followed by several episodes of pulmonary edema. A diagnosis of hyperthyroidism due to Graves' disease was confirmed by a laboratory test for thyroid function and anti-thyroid-stimulating hormone (TSH) receptor antibodies. The plasma concentration of thiamazole was measured before and at 1, 3, 4, 5, 6, and 24 h after administration of the drug. The dialyzability of the drug was investigated during hemodialysis therapy. On the non-dialysis day, the serum half-life of thiamazole (6.4 h) was similar to that in healthy subjects (4-6 h). Further, thiamazole was removed via the dialyzer during dialysis therapy. The initial dose of thiamazole was set at 15 mg/day for the patient. Free thyroid hormone levels began to decrease 2 weeks after the initiation of thiamazole, followed by the normalization of the values after 1 month. The patient's symptoms also subsided. Several confirmations of the concentration of thiamazole in the plasma in the morning on the first dialysis day of the week did not disclose a trend of accumulation in the blood. Although this is a single case report, it is suggested that thiamazole can be used for patients with ESRD. Careful monitoring of thyroid function, however, is recommended, because the intrathyroid action of thiamazole in uremia is unknown.