A case of superior sagittal sinus thrombosis is reported. The patient was a 27 years women and thrombosis occurred at 5 weeks pregnancy. This patient had one antithrombin III deficiency. Neurological feature associated hemiplegia and comatose with signs of severe intracranial hypertension (intracranial pressure greater than 50 mmHg). CT scan showed hemorrhagic infarct with man effect (real intracranial hematoma). This case was successful treated by surgical removal and low molecular weight heparin as anticoagulant. The patient recovered rapidly and kept only a slight focal deficit.