Candida species, including Candida dubliniensis, are a rare cause of meningitis. Herein, we report the second case of C. dubliniensis meningitis in a 49-year-old man with a history of hepatitis C virus-related cirrhosis, substance use disorder, and recent exposure to intravenous antibiotic therapy, presenting with confusion, abnormal gait, and urinary incontinence. Magnetic resonance imaging (MRI) of the brain showed marked hydrocephalus and leptomeningeal enhancement. Initial cerebrospinal fluid (CSF) studies were concerning for bacterial meningitis, although cultures were negative. Despite empiric treatment with broad-spectrum antibiotics, the patient’s mental status declined. The diagnosis of C. dubliniensis meningitis was not made until the third lumbar puncture. The patient was treated with liposomal amphotericin B and flucytosine. Despite improvement of hydrocephalus on MRI of the brain and sterilization of CSF, the patient’s mental status declined and he expired. This case highlights the difficulty in the diagnosis of C. dubliniensis meningitis as multiple lumbar punctures may be necessary. C. dubliniensis meningitis should be considered in the differential diagnosis for a patient with risk factors such as end-stage liver disease, human immunodeficiency virus infection, recent chemotherapy, substance use disorders, and recent broad-spectrum antibiotic use. A high index of suspicion is necessary as delay in initiation of therapy is associated with high mortality. The optimal treatment strategy has not been determined.