• Li Liu, Yushi Zhong, Bin Wu, Xianbi Tang, Ziwei Yi, and Chuzheng Pan.
• Department of Neurology, The First People's Hospital of Huaihua of University of South China, Huaihua, PR China.
• Medicine (Baltimore). 2022 Oct 14; 101 (41): e31088.

RationaleSpontaneous carotid cavernous fistula (CCF) is rare, and the expression of headache caused by it can be variable.Patient ConcernsA 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.DiagnosisDigital 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.InterventionsThe pseudoaneurysm was successfully treated with covered stent.OutcomesThe 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.LessonsHemicranial postural headache may be the first and characteristic sign of spontaneous CCF.Copyright © 2022 the Author(s). Published by Wolters Kluwer Health, Inc.

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