Many diseases may cause bilateral lower extremity edema. Diseases related to the heart, liver, and kidney shall be excluded first. The case of this study was a women aged 59 years with hyperthyroidism. The patient regularly took propylthiouracil (PTU) for 2 years. Bilateral lower extremity edema emerged as the first symptom, followed by hematuria, proteinuria, and impaired kidney functions. Initially, the medical team assumed the cause was glomerulonephritis with an unknown cause. After examination, the team discovered the patient was antineutrophil cytoplasmic autoantibodies (ANCA) positive. Through kidney biopsy, the team confirmed that the cause was PTU-related ANCA vasculitis. Therefore the delivery of PTU was discontinued, and steroid treatment was administered. The patient’s kidney functions gradually improved. In similar cases where diagnosis cannot be confirmed through symptom interpretations or general examinations, biopsy of the infected area should be performed to confirm the diagnosis of the cause. When drug-related ANCA vasculitis is confirmed, the delivery of medicine must be suspended immediately to avoid further lethal damage.