Abstract Candida parapsilosis is a very rare cause of meningitis. Though several cases have now been reported in neonates and children, only one has been described in an adult. We report on a 55-year-old male that was admitted due to altered mental status. He had recent sinus drainage and polypectomy, craniotomy with drainage of brain abscess, and ventriculo-peritoneal shunt placement. On admission, imaging studies showed no evidence of shunt dysfunction but did reveal extensive white matter decreased attenuation. Microscopic examination of the first 10 daily cerebrospinal fluid (CSF) cultures revealed yeast. Flucytosine and liposomal amphotericin B were started and externalization of shunt was performed on day 3. On day 8, CSF culture from admission grew C. parapsilosis; fluconazole was added. On day 10, daily CSF still showed yeast and cultures consistently grew C. parapsilosis. Shunt was removed and bilateral ventriculostomy drains were inserted. CSF after procedure as well as at follow-up examinations throughout his 3-month hospitalization were negative for yeast. Extended treatment with flucytosine and fluconazole was initiated. At 8-month follow-up, successful treatment of C. parapsilosis infection without recurrence was confirmed. This case underscores the need for suspicion of C. parapsilosis as a cause of meningitis after invasive surgeries in adults.