Hereditary antithrombin (AT) deficiency is an autosomal dominant inheritance disorder. Patients with AT deficiency have a high risk of thromboembolism, and intraoperative cardiopulmonary bypass management is difficult. We present the case of a 53-year-old man who was diagnosed with AT deficiency and who underwent an open heart operation for a right atrial hemangioma. We administered AT-III concentrate perioperatively to maintain his AT-III level up to 80% to establish cardiopulmonary bypass and prevent thromboembolic events postoperatively. The right atrial hemangioma was successfully excised after right atriotomy. The patient had an uneventful postoperative course and was discharged on postoperative day 14.
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