A case is reported of bilateral aspergillus endophthalmitis in an infant without any detectable systemic predisposition or focus of infection. The patient is the youngest recorded case of endogenous aspergillus endophthalmitis so far, with an onset of symptoms at the age of 15 days. After histopathological confirmation of the aetiology by enucleation of the atrophic eye, systemic amphotericin B therapy was instituted with excellent results in the fellow eye. This is the first recorded instance of a complete clinical cure in aspergillus ocular disease on systemic amphotericin B therapy alone.