Although blastomycosis of the central nervous system (CNS) occurs in simila
r to 4% of patients with blastomycosis, recurrent CNS blastomycosis is very
rare. We review the clinical features, treatment, and outcome of 4 previou
sly reported cases. We also report a case of recurrent CNS blastomycosis su
ccessfully treated with surgery and liposomal amphotericin B after an inade
quate response to amphotericin B therapy. This treatment may be an alternat
e approach for management of similar cases.