<b><i>Aim:</i></b> This study aimed to report a case of vitreous and optic nerve infiltration of a primary adrenal B lymphoma. <b><i>Case Report:</i></b> An 81-year-old patient consulted for decrease in vision in the left eye and pain with ocular movements during the last week. The patient was being treated with chemotherapy for an adrenal gland diffuse large B-cell lymphoma. On examination, vitritis was seen in both eyes and optic disc edema in his left eye. After an extensive study based on orbital and brain magnetic resonance imaging and vitreous cytology, an ocular infiltration by systemic B lymphoma was confirmed. A treatment based on intravitreal methotrexate was carried out, achieving the regression of the vitreous infiltration and resolution of the optic disc edema. Systemic B lymphoma metastasizing ocular structures is extremely infrequent. The manifestations may mimic an inflammatory disease. Ophthalmologists should be aware of these manifestations and consider among the masquerade syndromes.
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