LAUSR

Rationale: Spontaneous carotid cavernous fistula (CCF) is rare, and the expression of headache caused by it can be variable. Patient concerns: A case of a man hospitalized for high-intensity hemicranial headache which was aggravated by lying down and relieved when standing or sitting. The pain was of a pulsating character, localized on the right, behind the eye, followed by nausea and vomiting. He gradually appeared with ophthalmoplegia, decreased visual acuity and epistaxis. Diagnosis: Digital subtraction angiogram (DSA) showed a pseudoaneurysm arising from the internal carotid artery (ICA) that projected anteriorly and medially into the sphenoid sinus with occluded fistula. Interventions: The pseudoaneurysm was successfully treated with covered stent. Outcomes: The patient was then followed up clinically at the outpatient and seen in the outpatient clinic with no further episodes of nasal bleeding or new neurologic deficit. The vision loss and ophthalmoparesis were unchanged. Lessons: Hemicranial postural headache may be the first and characteristic sign of spontaneous CCF.