Invasive fungal infections are a significant cause of morbidity and mortality. Endogenous fungal endophthalmitis is a rare intraocular infection with potential vision threatening consequences. Our review of the literature revealed only one other case of Trichosporon endophthalmitis. Ocular fungal infections are difficult to eradicate because of the limited availability of systemic and intravitreal therapeutic agents and poor tissue penetration of current antifungals. Along with systemic antifungal agents, vitrectomy and intravitreal amphotericin B have been suggested as optimal treatments for fungal endophthalmitis. Other antifungals such as flucytosine and triazoles have recently received consideration. Although the current antifungal therapy is not highly successful, there remains a significant potential for more successful treatments in the future, based on the current studies. We report a case of endogenous trichosporon endophthalmitis that was successfully treated with micafungin and voriconazole. This combination has not been previously reported as a successful therapy in literature. More targeted research is required to uncover additional efficacious therapies to combat trichosporon.