Melioidosis is a disease of protean manifestations caused by the saprophytic gram-negative bacteria, Burkholderia pseudomallei. It ranges from asymptomatic infection to abscess, pneumonia, and disseminated disease. Here is an unusual case report of skull bone osteomyelitis due to melioidosis in a Human immunodeficiency virus (HIV) positive patient which was diagnosed on pus culture from the left parietal swelling that was drained after neurosurgical intervention. Appropriate antibiotic was started based on culture & sensitivity and finally patient improved clinically and radiologically.