The authors report on a case of intracranial candidiasis in an immunocompetent neonate with a ventriculo-peritoneal shunt. The child was known to be colonized with yeast as she had been treated for oral thrush; however, she did not have systemic candidiasis. Despite initial treatment with antifungal medication, intraventricular fungus balls developed that were visible on imaging and confirmed with pathological analysis. Multiple endoscopic intraventricular operations were required for excision of the initial and recurrent fungus balls, multiple fenestrations of loculations and cysts were performed, and ultimately 3 ventriculoperitoneal shunts were placed. The finding of an intraventricular fungus ball is a unique manifestation of intracranial candidiasis, and to the authors' knowledge has not been previously reported in the English literature.