Background: Septic embolism is a relatively common and potentially severe complication of infective endocarditis (IE), and most often are found in the brain, solid organs (e.g. spleen, kidney, lungs), and lower extremity. Interestingly, we found an acute venous thromboembolic event in IE patient with septicemia. Case illustration: A 42 -year -old male was referred from neighboring island to our hospital for IE and septic shock. On admission he complained of shortness of breath during heavy activity, with fatigue and fever for seven days. He also presented with acute onset of left upper extremity pain for three days. The pain was increased over time with burning sensation, exacerbated by activity, and there were no relieving factors. The left upper extremity looks bluish with bruising, accompanied by tenderness, and warm to touch. Laboratory examination showed leukocytosis (24.7 x 10 9 /L), thrombocytopenia (65 x 10 9 /L), elevated transaminase (SGOT 3.920 and SGPT 1.836), elevated serum creatinine (4.1 mg/dL), with an elevated infection marker (CRP 5.42 mg/L and procalcitonin 1.03 mu g/L). Echocardiography showed severe mitral regurgitation (MR) with 1.22 x 0.818 cm vegetation on the posterior leafl et, dilatation of left ventricle, and left atrium with a normal ejection fraction. The patient then underwent a duplex ultrasound examination of upper extremities and it revealed thrombus in left axillary vein until left brachialis vein, and no thrombus was detected in all upper extremity arteries level. Left sided infective endocarditis with acute venous thromboembolic (VTE) event in upper extremity was the most likely diagnosis. Therefore, empirical antibiotics and parenteral anticoagulant were administered during admission. Venous thromboembolic complication in left -sided IE is rare, but reported in several cases including VTE in central nervous system, pulmonary embolism, and lower extremity. Conclusion: Venous thromboembolic event in patients with IE is rare, but can occur in sepsis condition. In patients with known diagnosis of septic IE with acute presentation of swelling in extremities, VTE event should be suspected. Duplex ultrasound is an inexpensive, feasible, and producible examination for diagnosing VTE, which can direct us to determine further therapy namely anticoagulant therapy in IE.