Nonaneurysmal sulcal subarachnoid hemorrhage in a patient with atherosclerotic intracranial stenosis

CASE REPORT

Ana Gouveia1, Ana Inês Martins1, João Sargento-Freitas1-3, Luciano Almendra1, Fernando Silva1,2, Bruno Rodrigues1, Cristina Machado1, Gustavo Cordeiro1, and Luís Cunha1,3

1Neurology Department, Coimbra University and Hospital Centre, Coimbra, Portugal2Neurosonology Laboratory, Neurology Department, Coimbra University and Hospital Centre, Coimbra, Portugal3Faculty of Medicine of the University of Coimbra, Coimbra, Portugal

Correspondence: Ana Gouveia

Department of Neurology, Centro Hospitalar e Universitário de Coimbra

Praceta Mota Pinto 3000-075 Coimbra, Portugal

Email: anargouveia86@gmail.com

Background: Nonaneurysmal sulcal subarachnoid hemorrhage (sSAH) is a rare cause of cerebrovascular disease and rep-resents a small proportion of nontraumatic SAH.

Case report: We report the case of a 54-year-old male patient, who presented with severe abrupt headache, followed two days later by right visual field defect and dysphasia. Head CT revealed left peri-rolandic sSAH and two hypodense lesions (left cortical parieto-occipital and left subcortical parietal). Additional investigation found a severe MCA atherosclerotic stenosis.

Discussion: The case reported represents a rare cause of sSAH. We discuss possible pathophysiological mechanisms.

Keywords: Sulcal subarachnoid hemorrhage, Atherosclerotic disease, Intracranial stenosis

Citation: Gouveia et al. L. Nonaneurysmal sulcal subarachnoid hemorrhage in a patient with atherosclerotic intracranial stenosis. International Journal of Clinical Neurosciences and Mental Health 2016; 3(Suppl. 1):S13

DOI: http://dx.doi.org/10.21035/ijcnmh.2016.3(Suppl.1).S13

Published: 23 December 2016

© 2016 Gouveia et al. This is an open access article distributed under the Creative Commons Attribution License, which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited.

Open Access Publication Available at http://ijcnmh.arc-publishing.org

INTERNATIONAL JOURNAL OF

AND

CLINICAL NEUROSCIENCESMENTAL HEALTH

Special Issue on Controversies in Neurology. From the 10th World Congress on Controversies in Neurology (CONy), Lisbon, Portugal. 17–20 March 2016.

Abstract

Nonaneurysmal sulcal SAH due to atherosclerotic intracranial stenosis2

ARC Publishing

Introduction

Nonaneurysmal spontaneous sulcal subarachnoid hemor-rhage (sSAH) is defined as an hemorrhage located in the convexity of the brain, without involvement of adjacent parenchyma or extension into interhemispheric fissure, sylvian fissure, basal cisterns or ventricles. It is a rare cause of cerebrovascular disease and represents 5% of SAH [1]. We report the case of a patient with an exceptional cause of sSAH.

Case Report

A 54-year-old male patient presented with severe abrupt holocranial headache. Two days later, he noticed right visual field defect and fluctuating speech difficulties. Neurological examination revealed right homonymous hemianopia, dys-phasia, alexia and dyscalculia. The patient’s medical history included active smoking, hypertension and dyslipidaemia.

The initial head CT revealed left peri-rolandic sSAH and, at the fourth day, two new hypodense lesions (left cor-

tical parieto-occipital and left subcortical parietal). Brain MRI performed in the 7th day of symptoms confirmed the presence of two subacute ischemic lesions in the afore-mentioned locations (Figure 1).

Transcranial color coded Doppler (TCCD) evalua-tions were performed repeatedly during the first 10 days and found a persistent focal acceleration in left distal M1 segment of middle cerebral artery (MCA) [Lindegaard In-dex 4.5] with downstream flow attenuation. The digital subtraction cerebral angiography excluded the presence of aneurysms and arteriovenous malformations and showed a severe focal stenosis in left distal M1 segment of MCA, with a typical atherosclerotic morphology (Figure 2).

The patient was treated with high-dose statin and, after head CT excluding rebleeding, dual antiplatelet therapy.

Six months later, the neurological examination reveals right inferior homonymous quadrantanopia and dyscalcu-lia. There has been complete SAH reabsortion in the CT. In TCCD evaluation there is still evidence of severe left MCA stenosis.

Figure 1. Head CT (a, b, c): left peri-rolandic sSAH (a), left cortical parieto-occipital (b) and left subcortical parietal (c) lesions. Brain MRI (d – T2*, E - DWI, F - FLAIR) – sSAH (d) and subacute ischemic lesion (e and f ).

(a) (c)(b)

(d) (e) (f)

3Gouveia et al.

International Journal of Clinical Neurosciences and Mental Health 2016; 3(Suppl. 1):S13

Discussion

There are multiple known etiologies for sSAH, including vascular causes, namely, reversible cerebral vasoconstric-tion syndrome (RCVS)[2], cerebral amyloid angiopa-thy (CAA) [3], cerebral venous thrombosis [4], vascular malformations [5] and Moyamoya disease or syndrome [6]; and nonvascular causes as brain tumors [7], abscess-es [8] and coagulopathy [9]. The largest cohorts reported in literature suggest a difference in etiologies according to the age group. In patients younger than 60 years, RCVS is found more frequently, while in older patients CAA is more prevalent [10, 11].

Few cases of sSAH in patients with severe extracranial atherosclerotic disease have been reported, some of them in association with intracranial atherosclerotic disease [5]. Possible pathophysiological mechanisms are similar to that of Moyamoya disease. The severe arterial stenosis may trigger the development of fragile and dilated pial collater-

Figure 2. Transcranial color coded Doppler (A) persistent focal acceleration in left distal M1 segment of MCA. Digital subtraction cerebral angiography (B) - severe focal stenosis in left distal M1 segment of MCA, with a typical atherosclerotic morphology.

als in watershed zones that, under an acute hemodynamic change, can rupture and cause an sSAH.

The association of sSAH with isolated intracranial ath-erosclerotic stenosis is exceptional. Our patient had two ischemic lesions in watershed areas and a sSAH ipsilateral to the severe MCA stenosis. We believe the same patho-physiological mechanisms may have been implicated.

sSAH is distinct from most SAH. The etiologic investi-gation of a sSAH can be challenging due to its multiple pos-sible causes and their consequent therapeutic implications.

Abbreviations

CAA: Cerebral amyloid angiopathy; MCA: Middle cerebral artery; RCVS: Reversible cerebral vasoconstriction syndrome; SAH: subarach-noid hemorrhage; sSAH: Sulcal subarachnoid haemorrhage; TCCD: Transcranial color coded Doppler

Competing interests

The authors declare no conflict of interest.

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