A patient with a cholesteatoma secondary to chronic suppurative otitis media developed infective complications including cerebellar and cerebello-pontine abscesses. Burkholderia cepacia was isolated from ear swabs, infected tissue and surgical pus on five occasions over a six-month period. Despite exhibiting three different antibiotic susceptibility profiles, all five isolates were confirmed as clonally indistinguishable by pulsed-field gel electrophoresis. To the best of the authors' knowledge there are no previous reports of Burkholderia cepacia causing infection of the adult central nervous system.