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Introduction: To report a case of ocular dirofilariasis in Central Italy (Pisa, Tuscany) and review the previous cases of ocular dirofilariasis reported in Italy. Case description: A 67-year-old man complained about recurrent ocular redness and discomfort previously treated as conjunctivitis. The slit-lamp examination showed a round, translucent cystic swelling under the bulbar conjunctiva of the right eye, near the lateral canthus. Inside this lesion was observed the presence of a motile worm. A surgical procedure was immediately performed, excising a thin, segmented, greyish worm identified as Dirofilaria repens. After the excision, no recurrence of the symptomatology was recorded. Conclusion: Ocular dirofilariasis can lead to misdiagnosis due to its rare ocular manifestations and it is considered an emergent zoonosis in European countries. In Italy from 2001 to January 2018, seven new cases were reported in the literature. In our opinion, it is critical to record all the new cases to assess the epidemiological situation and increase the awareness of this rare infection.