Paracetamol anaphylaxis is a very rare event, with only a few cases described in literature and even less reported in children. We report the case of a 15-year-old boy, referred to Immunoallergy Department due to four reproducible episodes of anaphylaxis after paracetamol administration, since the age of 8 years. The most severe episode occurred at 12 years, characterized by glottis edema with respiratory distress, hypotension, generalized urticaria and facial edema, immediately after intravenous administration of paracetamol during a post-operatory recovery. He had always and still tolerates ibuprofen; an oral challenge test with meloxicam was negative. Skin prick and intradermal tests with paracetamol were negative. Serum-specific IgE and CAST to paracetamol were also negative. This report provides an alert to health-care professionals regarding the potential severity of reactions occurring within the therapeutic range of this widely used drug.