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Gülhane Askeri Tıp Akademisi, Göz Hastaklıkları Our aim is to describe an atypical case of primary antiphospholipid syndrome with bilateral retinal vein occlusions. A detailed clinical course with a 19-month follow-up is covered in the current case. The patient presented with retinal vein occlusion as a prevailing sign. He had hyperhomocysteinemia, methylene-tetrahydrofolate- reductase enzyme (MTHFR) C677T mutation and lupus anticoagulant (LA) seropositivity. He underwent several surgical interventions, and resulted in legal blindness in one eye. Ocular manifestations of primary antiphospholipid syndrome is variable, can be the first clinical sign, and result in legal blindness despite treatment. Keywords : Primary antiphospholipid syndrome, bilateral retinal vein occlusions; subdural hemorragia