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.