Stenotrophomonas maltophilia (S. maltophilia) is an emerging opportunistic bacterial pathogen with resistance to several commonly used antibiotics. Owing to its multidrug resistance (MDR), management of S. maltophilia blood stream infection (BSI) is challenging and requires the selection of appropriate antibiotic therapy. The presence of thrombocytopenia and shock are independent risk factors associated with increased mortality in patients with S. maltophilia BSI. We describe an unusual case of S. maltophilia BSI in a middle-age female complicated by dengue fever. We highlight the importance of early recognition of both dengue and S. maltophilia infection in management of such cases.