Intracranial infectious aneurysm (IIA) is one of the most severe complications of infective endocarditis (IE). Approximately 2-9% of patients with IE have IIA, which possibly results in severe neurological deficits. Currently, the most common treatment for IIA is endovascular treatment, while excision surgery is less common. The present study describes the case of a 33-year-old male patient who underwent a primary evacuation of an intracerebral hemorrhage (ICH) in the right temporal lobe. The patient was diagnosed with IE and IIA by examinations with an enhanced computed tomography scan, echocardiography and blood culture. In the recovery period after surgery, the patient suffered intracerebral rebleeding and underwent a surgery of IIA excision. The patient finally achieved a good prognosis without severe neurological dysfunction. In summary, IIAs are extremely rare entities, and a rare cause of spontaneous ICH. Previous research has demonstrated that the majority of ruptured IIAs receive endovascular treatment rather than conventional surgery. In the case presented herein, IIA surgical excision was successfully performed and complete pathological results were obtained, which has rarely been reported in the literature. The present case report reinforces the validity of traditional craniotomy according to the characteristics of IIAs.