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Melioidosis: an unusual cause of infective endocarditis: a case report. Introduction: As a causal organism in infective endocarditis, Burkholderia pseudomallei is rare. Burkholderia pseudomallei is intrinsically resistant to aminoglycosides but a gentamicin-susceptible strain was discovered in Sarawak, Malaysian Borneo in 2010. We report the first occurrence of infective endocarditis due to the gentamicin-susceptible strain of B. pseudomallei. Case presentation: A 29-year-old man presented with pneumonia and melioidosis septicaemia. His condition was complicated with infective endocarditis and septic emboli to the brain. Despite difficulties in reaching a diagnosis, the patient was successfully treated using intravenous gentamicin and ceftazidime and was discharged well. Discussion: The role of gentamicin in the treatment of the gentamicin-susceptible strain of B. pseudomallei remains unclear.