RATIONAL:: Infective endocarditis (IE) is defined as an infection of the endocardial surface of the heart, which may include one or more heart valves, the mural endocardium. PATIENT CONCERNS:: A 53-years-old man with a history of alcohol abuse was admitted in hospital for fever, paroxysmal atrial fibrillation cardioverted by Amiodarone and pulmonary infection. DIAGNOSIS:: A case of recurrent severe endocarditis, with neurological complications both ischemic and hemorrhagic and heart failure caused by Streptococcus agalactiae in healthy man we reported. INTERVENTIONS:: Surgery was performed 2 weeks after admission. OUTCOMES:: The onset of intracranial hemorrhage delayed second cardiac surgery and the patient died because of end-stage heart failure. CONCLUSIONS:: Infective endocarditis caused by S. agalactiae is very rare, particularly in patients without underlying structural heart disease. This study showed that IE due to S. Agalactiae is a disease with high mortality when associated with neurological complication, heart failure but especially when it is recurrent and hits valve prosthesis.