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Dirofilaria repens is the causative agent of human subcutaneous or, less often, ocular dirofilariasis. The work presents a rare case of ocular dirofilariasis manifested by previous subcutaneous migration accompanied by severe headache symptoms. In February 2017, a 58-yr-old man from Trnava region, western Slovakia, noticed red and itchy stripes on his left leg. Inflamed but painless stripes disappeared and showed up again every 5–7 days, migrating gradually towards the head. Approximately one month after the first skin´s alterations, strong pain in the left temple, with the swelling of the left face and the enlargement of mandibular lymph nodes appeared. Several days later, the patient felt excruciating pain of the right eyeball accompanied by strong nausea and subsequent vomiting. Ocular examination revealed the presence of a live worm in the subconjunctival space and morphological and molecular analyses of extracted helminth confirmed D. repens as etiological agent of the infection. According to clinical manifestation of the infection, it could be supposed that ocular form of the disease was the result of the migration of a parasite through the subcutaneous tissues. Moreover, a rare phenomenon of lymphadenitis of underlying lymph nodes and the swelling of left face accompanied the migration.