Burkholderia, a multidrug-resistant Gram-negative bacteria, is an uncommon cause of infection mostly in immunocompromised patients with a clinical profile very similar to tuberculosis. The most common conditions associated with this organism are cystic fibrosis and chronic granulomatous diseases. Bacteremia with it occurs in patients who are chronically ill and associated with significant morbidity and mortality. We are reporting here a case of perisplenic intra-abdominal abscess caused by Burkholderia cepacia in a patient with sickle cell disease (SCD).