The pathogenesis of Hashimoto’s encephalopathy has not been clearly elucidated and involvement of autoimmune damage has been proposed. We report a 23-year-old man who was emergently hospitalized for paroxysmal syncope and involuntary tremor with normal physical examination except for low heart rate. The patient was characterized by neurological symptoms, mild hypothyroidism, enlarged pituitary gland and extremely elevated thyrotropin, which all were reversed with levothyroxine alone. The case indicated that the pituitary-thyroid dysfunction may play a role in the pathogenesis of Hashimoto’s encephalopathy, hence, evaluation of pituitary gland should be recommended in diagnosis and treatment of Hashimoto’s encephalopathy.