We report a case receiving treatment with diclofenac sodium and ceftriaxone, who presented with facial unilateral angioedema attacks twice. Unilateral angioedema is a rare adverse reaction after NSAID usage. There is no previous report in the literature about ceftriaxone induced unilateral facial angioedema. To our knowledge this is the first reported case of unilateral facial angioedema following ceftriaxone treatment.