Cerebral venous sinus thrombosis (CVST) is a rare cause of stroke especially among young adults. The clinical presentation of CVST is diverse, which makes the diagnosis to be challenging. Lower cranial neuropathy is a rare presentation of CVST, with few cases in literature. We are describing a case of 39-year-old male patient, presented with left occipital neuralgia, with left lower cranial neuropathies due to left transverse and sigmoid sinus thrombosis; secondary to antiphospholipid syndrome, with good response to anticoagulation.