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Backgrounds/Aims: The authors describe the case of a 79-year-old Caucasian woman who presented an ocular adnexal lesion as the first clinical manifestation of a systemic follicular lymphoma, highlighting the clinicopathological features of this rare entity and its potential to be misdiagnosed as marginal zone lymphoma of the mucosa-associated lymphoid tissue. Methods: Conjunctival impression cytology was performed for a rapid initial diagnosis of lymphoma, and subsequent histopathological and immunohistochemical studies were carried out for its confirmation and to identify the best therapeutic regimen. Results: After the initial presentation and diagnosis, she was submitted to complete clinical evaluation; confluent retroperitoneal lymphadenopathy was detected through abdominal computed tomography, characterizing clinical stage III. Conclusion: Awareness of this lymphoma is important when making a diagnosis of ocular adnexal lymphoid neoplasms for its appropriate evaluation and management.