Voriconazole (VRC) has not previously been reported to cause angio-oedema. Here, we report a case of angio-oedema associated with VRC therapy. A 37-year-old woman with relapsing invasive vertebral aspergillosis received intravenous VRC and developed angio-oedema 10 days after starting therapy. This condition rapidly diminished after administration of intravenous antihistaminics and did not necessitate cessation of VRC treatment. The treatment was continued for 6 months without recurrence of the symptoms. After 18 months, the patient was in good health. To our knowledge, this is the first report of an angio-oedema associated with VRC.