Coxiella burnetii is one of the most common causes of blood culture-negative infective endocarditis (IE). However, only a few cases of cardiac implantable electronic devices (CIED) infection have been reported in the literature. Herein, we present a case of CIED-related blood culture-negative infection attributed to C. burnetii. A 54-year-old male was admitted to our hospital due to prolonged fatigue, a low-grade fever lasting more than a month, and weight loss. Three years ago, he received an implantable cardiac defibrillator (ICD) as a primary prevention measure against sudden cardiac death. An initial transthoracic and transesophageal echocardiography showed a dilated left ventricle with severely impaired systolic function, while the ventricular pacing wire was inside the right ventricle with a large echogenic mass (2.2 × 2.5 cm) adherent to it. Repeated blood cultures were negative. The patient underwent transvenous lead extraction. A transesophageal echocardiography after the extraction revealed multiple vegetations on the tricuspid valve with moderate to severe valve regurgitation. A surgical replacement of the tricuspid valve was determined after a multidisciplinary heart team approach. Serology tests showed increased IgG antibodies in phase I (1:16,394) and phase II (1:8192), and a definite diagnosis of CIED infection was made based on the serological tests.