To report a rare case of Aspergillus endophthalmitis as a preceding symptom of central nervous system (CNS) lymphoma.

A 66-year-old female was admitted to our clinic with mental change for 3 days. Seven months earlier, she had been diagnosed with retinal vasculitis in an ophthalmology clinic because of blurred vision in both eyes and was administered steroid therapy. Three months earlier, because of progressive symptoms, vitreous fluid culture had been performed and showed Aspergillus endophthalmitis. She was treated with intravitreous voriconazole injection and oral voriconazole. Initial brain magnetic resonance imaging (MRI) and cerebrospinal fluid study was normal. Two months later, a second MRI showed multiple enhancing lesions, which were aggravated on the third MRI at admission to our clinic. Although brain biopsy was not performed due to the poor condition of the patient, CNS lymphoma was suspected based on the neuroimaging. After steroid pulse therapy and whole brain radiation, follow-up neurologic examination showed improved mental state, and follow-up MRI showed remarkable shrinkage of multiple lesions.

As Aspergillus endophthalmitis is an opportunistic infection in those with an immune-compromised state and the orbit is near the central nervous system, the clinician should be alert to concomitant disorders in CNS. For a prompt and accurate diagnosis of CNS disorder, early evaluation of neurologic symptoms beyond symptoms of endophthalmitis and neuroimaging is essential.