A 49-year-old woman treated with increasing dosage of propylthiouracil (PTU) in order to control hyperthyroidism, developed progressive necrotic ulcers in the oral cavity, oropharynx and rhinopharynx and vasculitic ulcers on both auricular pinnae after a few days. The PTU treatment was immediately discontinued and the mucosal and skin manifestations resolved promptly. Laboratory findings, including anti-neutrophil cytoplasmic antibodies (ANCA), suggested allergic vasculitis. This is to our knowledge the first reported case of oral manifestations of PTU-induced allergic vasculitis.