Purpose: To describe a rare case of odontogenic brain abscess. Materials and methods: A healthy, 35-year-old male had two dental implants placed in a simultaneously augmented maxillary sinus. One implant failed and the patient developed a maxillary sinusitis that failed to improve following antibiotic treatment at home. The neglected sinus infection led to formation of a brain abscess. The patient was hospitalised only when he had pan sinusitis with neurological signs. Symptoms were headache attacks, a subfebrile fever and a purulent secretion from the left nostril. The osteomeatal complex was blocked, the maxillary sinus was filled with pus and the Schneiderian membrane thickened. The patient was treated with intravenous antibiotic treatment. Computerised tomography (CT) and magnetic resonance imaging (MRI) scans and functional endoscopic sinus surgery (FESS), were implemented. When his conditions worsened, the patient underwent a left frontal mini craniotomy. Results: Following the craniotomy and antibiotic treatment, there was a gradual resolution and the patient was dismissed after 2 months in hospital with no neurological deficit or signs of sinusitis. Conclusions: Maxillary sinusitis following dental implant insertion and concomitant maxillary sinus elevation should be treated immediately and thoroughly since untreated sinusitis may cause life-threatening situations such as a brain abscess. In case of severe infection, clinicians should refer immediately the patient to hospital specialists. Keywords: brain abscess, dental implant, maxillary sinus augmentation Conflict-of-interest statement: The authors have stated explicitly that there are no conflicts of interest. The manuscript was self-funded.