Purpose: Cryptococcus neoformans is a budding yeast that has a tropism for the central nervous system where it may cause meningitis, which has a high mortality rate. Endophthalmitis is rare and typically occurs in the setting of meningitis and disseminated disease. This report describes an atypical presentation of cryptococcal endophthalmitis and outlines the appropriate workup and management for this disease. Methods: A case report is presented. Results: A 66-year-old man on chemotherapy who presented with blurry vision without any extraocular symptoms was diagnosed with cryptococcal endophthalmitis and found to have underlying bloodstream and central nervous system infection on subsequent workup. He was treated with intravenous and intravitreal antifungals and remained systemically well, although a large subretinal mass lesion remained stable at 2-month follow-up. Conclusions: Cryptococcal endophthalmitis may be the initial presentation of disseminated cryptococcal disease. If vitreous cultures grow Cryptococcus, clinicians should presume that meningitis is present, regardless of symptoms, until proven otherwise.