This article reports a case of a 38-year-old female who developed drug hypersensitivity syndrome one month after receiving propylthiouracil. The patient showed improvement with adalimumab, corticosteroids, intravenous immunoglobulin, and plasma exchange. Propylthiouracil is a rare medication associated with drug hypersensitivity syndrome. Additionally, during the follow-up after discharge, rapid changes in thyroid autoantibodies and thyroid function were observed in this patient. By analyzing the progression of this case and reviewing literature, it aims to enhance clinical understanding and management.