AIM: To report a rare case of melioidosis presenting as orbital abscess, who was successfully treated with high dose of intravenous ceftazidime. METHODS: A case report. RESULTS: A 55-year-old Malay gentlemen who was newly diagnosed with diabetes mellitus, presented with prolonged low grade fever for three weeks and left eye swelling for five days duration. Initial CT scan of brain and orbit showed left periorbital cellulitis and acute left sphenoidal sinusitis. Initial swab culture grew Pseudomonas sp. His general condition improved with regular antibiotics. However, upon completion of intravenous therapy his condition worsened and the left eye became more proptosed. Repeat CT scan of the brain and orbit showed left eye orbital abscess with intracranial exten-sion. Swab culture from fistula of the lateral part of upper eyelid showed Burkholderia pseudomallei. He was treated with high dose of intravenous ceftazidime, oral co-trimoxazole for the acute management and on maintenance dose of oral co-trimoxazole for 2 months. He responded well to treatment and had no relapse up to one year post treatment. Unfortunately his left eye vision was not salvageable. CONCLUSION: This case illustrates a rare presentation of orbital abscess due to melioidosis which was complicated with cerebral abscess and septicemia. An accurate diagnosis was essential and high dose of susceptible antibiotics was important for the institution of therapy to successfully treat this potentially fatal condition.