A term male newborn (9 days old) without significant perinatal risk factors was initially admitted for hyperbilirubinemia and later developed septic shock with focal neurological signs. Examination revealed fontanelle collapse, poor reflexes, dry mucous membranes, right ptosis, abnormal pupillary reflexes, exotropia, and corneal reflex loss. Additional signs later appeared, including facial asymmetry, mydriasis, limited eye movements, and nystagmus. Despite negative CSF and blood cultures, a CT scan showed occipital edema, white matter changes, and sinus confluence thrombosis. Laboratory tests for inherited thrombophilias were negative and the patient was treated with enoxaparin before transitioning to rivaroxaban for discharge. Severe visual impairment was later diagnosed, and the patient remains under observation for further neurological sequelae.

This case highlights the critical importance of recognizing CVST with focal neuro-ophthalmological signs. Early anticoagulation and close monitoring are essential to prevent complications and improve outcomes in neonatal CVST.