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Aspergillus and Acanthamoeba cause central nervous system (CNS) infection mostly in immunocompromised hosts, disseminating via the hematogenous route from a primary focus in the respiratory tract. We are presenting a case of an immunocompetent patient diagnosed with Aspergillus and Acanthamoeba coinfection of the CNS, a rare finding. A positive cerebrospinal fluid (CSF) galactomannan antigen test confirmed the presence of Aspergillus, and direct visualization in CSF sample and culture confirmed the presence of Acanthamoeba.
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