Dados do Trabalho

Título

Unusual presentation of ischemic stroke due to stenosis of persistent primitive hypoglossal artery: a case report

RESUMO

Case presentation: A 74-year-old man only treated for benign prostatic hyperplasia underwent elective magnetic resonance imaging (MRI) to investigate left hypoacusis with months of evolution. An area of bilateral parieto-occipital diffusion restriction was detected with hypersignal in fluid-attenuated inversion recovery and hyposignal in the apparent diffusion coefficient. The patient was admitted without any neurological deficits, but reported a recent episode of vertigo lasting seconds in addition to reduced visual acuity. He had a blood pressure of 129/74mmHg and capillary blood glucose level of 97mg/dL. Therefore, a dual antiplatelet therapy was initiated. During the investigation, carotid and vertebral Doppler revealed non-significant carotid stenosis. Electrocardiography revealed sinus rhythm and Holter monitoring detected minimal changes. Transthoracic echocardiography identified an enlarged left atrium and ventricle with concentric remodeling. Computed tomography angiography revealed bilateral vertebral artery (VA) hypoplasia and a persistent primitive hypoglossal artery (PPHA) originating from the right internal carotid artery, giving rise to the basilar artery, in addition to a hypodense plaque during its course in the hypoglossal canal, reinforced by vessel wall MRI. Craniocervical junction computed tomography did not reveal any extrinsic compression.
Discussion: PPHA is a rare vascular anomaly that is usually detected incidentally during cerebral angiography and is generally asymptomatic. It is mainly associated with aneurysms, probably because of the anomalous structure of the vessel wall, which can expose the basilar trunk to excessive hemodynamic stress. As most patients have hypoplastic VA, the blood supply to the posterior circulation travels through PPHA. Therefore, in ischemic stroke patients with PPHA, both the anterior and posterior circulations are often involved. We present the case of a patient with PPHA and bilateral hypoplasia of the VA, who presented with a bilateral oligosymptomatic stroke with a hemodynamic component caused by a plaque found in the hypoglossal artery, which is an uncommon location of stenosis.
Final comments: We demonstrate a case of rare anatomic variation communicating the anterior and posterior circulations. In this way, we show the importance of knowing the anatomical variations in cerebral circulation to identify possible unusual presentations of stroke and to choose the best secondary prevention.

Palavras Chave

Stroke; artery stenosis; persistent primitive hypoglossal artery.