BACKGROUND Pseudomonas luteola (P. luteola) is a Gram-negative, oxidase-negative is an environmental organism that is isolated from soil, water, and damping areas, and is rarely found as a human pathogen. Most infections in which P. luteola is implicated are associated with a breach in immune barrier, such as indwelling catheters, prosthetic devices, immunocompromised conditions and surgical wounds. CASE REPORT A 9 years old girl, known case of Chron’s disease and recurrent urinary tract infections on prophylactic antibiotics, presented with an acute-onset abdominal pain associated with fever reaching 40C and vomiting. She was placed on a peripherally inserted central line for total parenteral nutrition and developed sepsis on the 30th day of admission. Septic workup revealed P. letuola infection with right atrial vegetation. Treatment with Piperacillin/tazobactam yielded an uneventful recovery. CONCLUSIONS P. luteola can be involved in serious infections in susceptible individuals. Serious outcomes may be associated with infective endocarditis especially on a background of valvular prosthesis and central lines. The definitive treatment of catheter related infective endocarditis caused by P. leuteola is the removal of the lines along with an appropriate antibiotic regimen-based AST result.