Aspergillus infections in lung transplant patients are frequently reported with a large pattern of manifestations varying from simple colonization of the lungs to complicated infections. Pulmonary invasive aspergillosis and disseminated aspergillosis often result in death. The majority of cases occur during the first months after transplantation with pulmonary involvement and have been described as the first clinical localization of the disease. Here we present the first reported case of an endophthalmitis caused by Aspergillus fumigatus developing 18 months after lung transplantation, and presenting as a manifestation of invasive aspergillosis.