Background: Melioidosis is a serious infection caused by soil-dwelling Gram-negative bacillus Burkholderia pseudomallei. It is most commonly reported in Northern Australia, Southeast and Southern Asia, China, and Taiwan. Methods: A case report and short review of the literature are presented. Presentation, diagnosis including genomic sequencing, and acute and long-term management are discussed. Results: A 58-year-old female presented with chronic rhinosinusitis secondary to melioidosis. This is the third reported incidence of sinusitis secondary to melioidosis, which occurred in an otherwise well female with no risk factors and no apparent cause of exposure. Treatment involved an acute phase in which meropenem was administered parenterally for two weeks, followed by a prolonged oral course of trimethoprim-sulfamethoxazole for three months, as per recommended guidelines. Conclusion: In patients presenting with refractory chronic rhinosinusitis, ENT surgeons should consider the presence of unusual causative pathogens such as B pseudomallei, particularly in those with recent travel history to Northern Queensland and/or Southeast Asia.