Amphotericin B resistance among isolates of Candida lusitaniae has distinguished it among Candida species. Because no comprehensive review has been published recently, we provide a case report and a literature review of C lusitaniae infection to update and better characterize the illness in the era of azole availability and standardized methodologies for antifungal susceptibility testing. C lusitaniae infection in the 55 cases surveyed in this review occurred in relatively young patients (median age, 44 years). Fungemia was found in 80% of patients. Other infection syndromes, including peritonitis, meningitis, and urinary tract infection, were much less common. Three-fourths of the patients had serious underlying medical conditions. Despite the presence of fungemia and predisposing comorbidities, death due to C lusitaniae infection was uncommon among treated patients (5.00%). Moreover, in vitro susceptibility testing results for amphotericin B did not appear to predict patient outcome in this survey.