Journal of Clinical Neuroscience xxx (2018) xxx–xxx Contents lists available at ScienceDirect Journal of Clinical Neuroscience journal homepage: www.elsevier.com/locate/jocn Case report Vertebrobasilar dolichoectasia presenting simultaneously with ischemic and hemorrhagic strokes Tia Chakraborty a, Giuseppe Lanzino b, Kelly D. Flemming a,⇑ a b Mayo Clinic Department of Neurology, Mayo Clinic, Rochester, MN, USA Mayo Clinic Department of Neurosurgery, Mayo Clinic, Rochester, MN, USA a r t i c l e i n f o Article history: Received 30 July 2018 Accepted 24 October 2018 Available online xxxx Keywords: Vertebrobasilar dolichoectasia Ischemic stroke Intracerebral hemorrhage Fusiform aneurysm Magnetic resonance imaging a b s t r a c t Background: Vertebrobasilar dolichoectasia (VBDE) can present with brainstem- compressive symptoms, cerebral ischemia, or hemorrhage. Herein we describe a unique patient with VBDE who presented with hemorrhage and cerebral ischemia simultaneously. Case: Patient is a 76-year-old man presenting with an acute, symptomatic left pontine ischemic stroke heralded by ataxic dysarthria and right ataxic-hemiparesis. MRI scan of the brain demonstrated acute cerebral ischemia in the left pons, a small right thalamic hemorrhage, and evidence of VBDE. Conclusion: The perforating vessels branching off of the pathologic VBDE may result in both ischemia and hemorrhage. This is the first known case, to our knowledge, in which hemorrhage and cerebral ischemia occurred simultaneously. Ó 2018 Elsevier Ltd. All rights reserved. 1. Case description A 76-year-old man with a history of hyperlipidemia and mild cognitive impairment presented with acute dysarthria, incoordination, and gait ataxia. He was initially hypertensive at 200/123 mm Hg. CT head demonstrated acute intracerebral hemorrhage (ICH) in the right thalamus, a chronic infarct in the left thalamus, and diffuse intracranial dolichoectasia (Fig. 1 Row A). Neurologic examination was significant for ataxic dysarthria and mild right ataxic hemiparesis. His gait was also ataxic. MRI of the brain revealed an acute ischemic stroke (AIS) in the left pons likely responsible for patient’s presenting symptoms. In addition, there were chronic infarcts in bilateral thalami and a 5 mm ICH in the right thalamus (Fig. 1 Row B). The crosssectional diameter of the basilar artery was 10 mm. The basilar tip indented the floor or the 3rd ventricle without resultant hydrocephalus. MRA head confirmed vertebrobasilar dolichoectasia (VBDE) in addition to dolichoectasia of the supraclinioid carotid arteries. Additional evaluation was unremarkable for a cardiac source of the ischemic stroke. Thus, the cerebral ischemia was attributed to small-vessel disease ultimately related to the VBDE. Similarly, the small, deep hemorrhage was attributed to perforating artery ⇑ Corresponding author at: Department of Neurology, Mayo Clinic, 200 First Street SW, Rochester, MN 55905, USA. E-mail address: Flemming.Kelly@mayo.edu (K.D. Flemming). friability related to parent-vessel pathology. As VBDE can be associated with diffuse vascular disease extracranially [1,2], an aortic ultrasound was pursued and demonstrated a 3.1 cm fusiform aneurysm of the abdominal aorta. Management comprised of resuming a full-dose daily aspirin 7 days following hemorrhage, blood pressure control, and initiating statin therapy. Due to the diffuse nature and length of the VBDE and the patient’s recent ischemic stroke, no endovascular intervention was recommended. 2. Discussion VBDE is a condition characterized by ectasia (enlargement) and tortuosity of the vertebrobasilar system [3,4]. Patients with VBDE may present with ischemic stroke, hemorrhage, or brainstemcompressive symptoms, or VBDE may be an incidental finding [1,3,4]. We report the first case, to our knowledge, of a patient presenting with both cerebral ischemia and intracranial hemorrhage simultaneously leading to a dilemma in medical management. Proposed mechanisms of cerebral infarction in patients with VBDE include low flow, thromboembolism, dissection, or compression of perforating vessels [2,5–7]. Some have proposed that the elongation and tortuosity of the vertebrobasilar arteries can stretch the perforating branches leading to reduced flow as a mechanism as well [8,9]. In addition, Pico and colleagues demonstrated that VBDE was independently associated with small-vessel, lacunar strokes [9]. Such is the case with our patient, who had a small vessel infarct in the pons. https://doi.org/10.1016/j.jocn.2018.10.092 0967-5868/Ó 2018 Elsevier Ltd. All rights reserved. Please cite this article in press as: Chakraborty T et al. Vertebrobasilar dolichoectasia presenting simultaneously with ischemic and hemorrhagic strokes. J Clin Neurosci (2018), https://doi.org/10.1016/j.jocn.2018.10.092 2 Case report / Journal of Clinical Neuroscience xxx (2018) xxx–xxx Fig. 1. VBDE with Stroke. Row A: CT head showing vertebrobasilar dolichoectasia as well as ectasia of the right supraclinoid internal carotid artery, intracerebral hemorrhage in right thalamus (arrow), and a chronic left thalamic ischemic stroke (arrowhead). Row B: T2 MRI sequences demonstrate similar findings as the CT as well as a new infarct in the left pons (arrow). Hemorrhage can occur due to VBDE and either result in subarachnoid hemorrhage (SAH) due to rupture of the large vessel (e.g., basilar artery), or intracranial hemorrhage (ICH) due to diseased perforating vessels off the main large vessel [1,3,10,11]. Pathology of VBDE that make these vessels vulnerable to hemorrhage includes fragmentation of the internal elastic lamina, thinning of the media, and areas of chronic dissection [12]. In smaller vessels, arteriolar damage may be due to parent-vessel pathology in addition to hypertensive degeneration. In the case of VBDE, ICH commonly occurs in the pons, thalamus, or midbrain [10,11]. Thus, we believe the right thalamic small, deep hemorrhage was also due to the VBDE. We theorize that this friability of perforating vessels off the basilar artery and posterior cerebral artery in a patient with VBDE resulted in simultaneous pontine cerebral ischemia and thalamic ICH. This simultaneous presentation with hemorrhage and ischemia posed management dilemmas. Given that recurrent stroke is a greater risk than bleeding in patients with VBDE [1,3,11] and the patient’s bleed was intracranial due to a perforating vessel rupture, rather than subarachnoid, we opted to initiate medical therapy aimed at reducing future ischemic stroke risk. Author contributions and disclosures Tia Chakraborty, M.D., authored, conceptualized, acquired data, drafted, and revised the manuscript. She had full access to all of the data in the study and takes responsibility for the integrity of the data and the accuracy of the data analysis. She reports no disclosures or conflict of interest. Kelly Flemming, M.D., conceptualized revised the manuscript. She reports no disclosures or conflict of interest. Giuseppe Lanzino, M.D., conceptualized and revised manuscript. He reports no disclosures or conflicts of interest. This research did not receive any specific grant from funding agencies in the public, commercial, or not-for-profit sectors. CRediT authorship contribution statement Tia Chakraborty: Methodology, Data curation, Writing - review & editing. Giuseppe Lanzino: Writing - review & editing. Kelly D. Flemming: Conceptualization, Data curation, Methodology, Writing - review & editing. References [1] Flemming KD, Wiebers DO, Brown Jr RD, et al. The natural history of radiographically defined vertebrobasilar nonsaccular intracranial aneurysms. Cerebrovasc Dis 2005;20:270–9. https://doi.org/10.1159/000087710. [2] Lou M, Caplan LR. Vertebrobasilar dilatative arteriopathy (dolichoectasia). Ann N Y Acad Sci 2010;1184:121–33. https://doi.org/10.1111/j.17496632.2009.05114.x. [3] Flemming KD, Wiebers DO, Brown Jr RD, et al. Prospective risk of hemorrhage in patients with vertebrobasilar nonsaccular intracranial aneurysm. J Neurosurg 2004;101:82–7. https://doi.org/10.3171/jns.2004.101.1.0082. [4] Smoker WR, Corbett JJ, Gentry LR, Keyes WD, Price MJ, McKusker S. Highresolution computed tomography of the basilar artery: 2. Vertebrobasilar dolichoectasia: clinical-pathologic correlation and review. AJNR Am J Neuroradiol 1986;7:61–72. [5] Ince B, Petty GW, Brown Jr RD, Chu CP, Sicks JD, Whisnant JP. Dolichoectasia of the intracranial arteries in patients with first ischemic stroke: a populationbased study. Neurology 1998;50:1694–8. [6] Nishizaki T, Tamaki N, Takeda N, Shirakuni T, Kondoh T, Matsumoto S. Dolichoectatic basilar artery: a review of 23 cases. Stroke 1986;17:1277–81. [7] Pessin MS, Chimowitz MI, Levine SR, et al. Stroke in patients with fusiform vertebrobasilar aneurysms. Neurology 1989;39:16–21. [8] Pico F, Labreuche J, Touboul PJ, Amarenco P, Investigators G. Intracranial arterial dolichoectasia and its relation with atherosclerosis and stroke subtype. Neurology 2003;61:1736–42. [9] Pico F, Labreuche J, Touboul PJ, Leys D, Amarenco P. Intracranial arterial dolichoectasia and small-vessel disease in stroke patients. Ann Neurol 2005;57:472–9. https://doi.org/10.1002/ana.20423. [10] Passero SG, Calchetti B, Bartalini S. Intracranial bleeding in patients with vertebrobasilar dolichoectasia. Stroke 2005;36:1421–5. https://doi.org/ 10.1161/01.STR.0000172311.64662.9c. [11] Passero SG, Rossi S. Natural history of vertebrobasilar dolichoectasia. Neurology 2008;70:66–72. https://doi.org/10.1212/01. wnl.0000286947.89193.f3. [12] Mizutani T, Miki Y, Kojima H, Suzuki H. Proposed classification of nonatherosclerotic cerebral fusiform and dissecting aneurysms. Neurosurgery 1999;45:253–9. discussion 9–60. Please cite this article in press as: Chakraborty T et al. Vertebrobasilar dolichoectasia presenting simultaneously with ischemic and hemorrhagic strokes. J Clin Neurosci (2018), https://doi.org/10.1016/j.jocn.2018.10.092