Background: Blood culture-negative infective endocarditis (BCNIE) refers to infective endocarditis (IE) with negative microorganism growth using usual blood culture method. It poses dilemma in terms of diagnosis, associated complication, and treatment. The study aims to highlight the occurrence of acute bilateral lower limb ischemia in BCNIE and its management approach. Case presentation: We present a BCNIE patient who developed acute bilateral lower limb ischemia, Rutherford stage 2b despite on course of antibiotic treatment. Patient underwent emergency bilateral open femoral embolectomy and right profundaplasty. A full six-weeks course of antibiotics and short-term anticoagulant successfully reverse the acute ischemic event. Conclusion: This case highlighted that despite the rarity of BCNIE causing limb ischemia, it can still occur due to significant risk factors. The early recognition of acute limb ischemia and prompt treatment are deemed important in saving the threatened limb from being amputated.

Keywords: endocarditis, embolectomy, limb salvage, mitral valve insufficiency, thromboembolism.