Antithyroid drug therapy with propylthiouracil (PTU) is commonly used in patients with Grave's disease. Common side effects of PTU include leukopenia, agranulocytosis, skin rash and abnormal liver function tests. Rarely, it can induce anti-neutrophil cytoplasmic antibody (ANCA) associated vasculitis which is a serious adverse effect during PTU therapy for Grave's disease. Withdrawal of PTU and the administration of corticosteroids ameliorates these manifestations. A case of propylthiouracil induced vasculitis is being reported here.