We present the case of a 38-year-old male patient with a history of recurrent ischemic cerebrovascular disease without determined etiology and venous thrombosis in lower limbs. Due to the aetiological diversity of cerebral infarction in a young adult, he underwent a series of clinical tests, which resulted in the diagnosis of a primary antiphospholipid syndrome associated with hyperhomocysteinemia.
Once the diagnosis was made, he was given therapy with anticoagulants and corticoid pulses; with posterior improvement. Antiphospholipid syndrome is part of the differential diagnosis in young women with cerebral infarction; most cases have been reported in its secondary form, but finding it in primary form and in a male patient is rare. Also, increased homocysteine values are related to the severity of the first cerebrovascular event, but not to recurrent events.