• Sen He, Ming-Li Wei, Fei Xie, and Seidu A Richard.
• Department of Neurosurgery, The First People's Hospital of Ziyang City, No.66 Rende West Road, Ziyang, PR China.
• Medicine (Baltimore). 2021 Aug 13; 100 (32): e26904e26904.

RationalePersistent primitive hypoglossal artery (PPHA) is a rare and permanent carotid-vertebrobasilar anastomoses. Patients with PPHA usually have higher changes of developing intracranial aneurysms due the high intracranial hemodynamics. Although cases of PPHA alone and PPHA with aneurysms have been reported in literature, cases of fenestrated PPHA harboring a ruptured aneurysm have seldomly be reported in literature. We present a rare occurrence of a fenestrated PPHA harboring a reputed aneurysm.Patients ConcernsA 43-year-old woman was presented with a sudden-onset severe headache and nausea.DiagnosisComputerized tomography scan showed third, fourth, and bilateral ventricular hemorrhages. Computed tomographic angiogram showed a PPHA with fenestration malformation and a cystic protrusion consistent with an aneurysm.InterventionThe patient underwent a successful stent-assisted coil embolization via the trans-arterial route under general anesthesia.OutcomesTwo years follow-up revealed no recurrence of her symptomatology and she is currently well and go about her normal daily life.ConclusionFenestrated PPHAs harboring aneurysms may be more prone to rupture because of the fenestration and connective tissue weakness of the artery as well as changes in hemodynamics of the already malformed and weak artery.Copyright © 2021 the Author(s). Published by Wolters Kluwer Health, Inc.

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