Pseudozyma spp. are described as environmental yeasts but have also been identified as rare human pathogens found in immunocompromised patients. This systematic review details the clinical manifestations, diagnostic methodology, and empirical anti-fungal therapy for this rare yeast. PubMed, LILACS, Scielo, and Web of Science databases were searched for articles about Pseudozyma spp. infections from inception to June 2019. Inclusion criteria were any published studies that included patients with Pseudozyma spp. infection. Infections were identified using criteria set forth by the European Organization for Research and Treatment of Cancer, and were further classified according to clinical, laboratory, or radiologic findings, microbiologic confirmation, and response to therapy. Eleven articles were included with 15 patients. Oncological and/or hematological disorders were the most reported risk factors. Nontraditional microbiological methods correctly identified Pseudozyma spp., whereas traditional methods failed to identify fungal genus. Species were identified by sequencing, and most demonstrated a higher minimal inhibitory concentration (MIC) for fluconazole and echinocandins. MICs for itraconazole, voriconazole, and posaconazole varied by species. All isolates were susceptible to amphotericin B, which was the most used treatment. Pseudozyma spp. infections usually present with fever and are diagnosed by blood culture. Most species studied appeared to be resistant to fluconazole and echinocandin. Voriconazole, posaconazole, and amphotericin were effective in treating P. aphidis. However, more studies are needed to evaluate voriconazole and posaconazole in species other than P. aphidis.

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