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Kavin Vanikieti, Piyaphon Cheecharoen, Panitha Jindahra, Sirin Lueangaram, Tanyatuth Padungkiatsagul
Vertebrobasilar dolichoectasia (VBD) is a rare dilative arteriopathy defined as elongation or widening of the intracranial vertebral and/or basilar arteries. The prevalence ranges from 0.06% to 5.8%. The majority of VBDs are asymptomatic. Downbeat nystagmus has been reported as a component of an infrequent ocular movement disorder in VBD. Nevertheless, oculopalatal tremor (OPT), delayed sequelae of a brainstem lesion, has never been demonstrated in VBD cases. Synchronized rhythmic involuntary contractions of the soft palate with an ocular pendular nystagmus, predominantly vertical pendular nystagmus, are the hallmark presentation...
2016: International Medical Case Reports Journal
Vicente Vanaclocha, Juan Manuel Herrera, Deborah Martínez-Gómez, Marlon Rivera-Paz, Cristina Calabuig-Bayo, Leyre Vanaclocha
BACKGROUND: surgical treatment of trigeminal neuralgia (TN) associated with vertebro-basilar dolichoectasia (VBD) is challenging. AIM: to analyze the treatments for this disease discussing its advantages and drawbacks, presenting our own technique and series. METHODS: retrospective study January 2006-January 2016. On pre and postop MRI images VBD deviation from midline, BA and VA diameter, and BA apex distance above posterior clinoid process were measured...
August 29, 2016: World Neurosurgery
Kyeong Joon Kim, Jong-Min Kim, Yun Jung Bae, Hee Joon Bae, Beomseok Jeon, Jae Hyung Kim, Jeong Ho Han, Chang Wan Oh
BACKGROUND: Hemifacial spasm (HFS) is frequently caused by vascular compression of the facial nerve. Vertebrobasilar dolichoectasia (VBDE) may cause vascular crowding in the limited space of the posterior fossa, increasing the chance of vascular compression of the facial nerve. We investigated the prevalence of VBDE in HFS. METHODS: We analyzed the presence of VBDE on 3.0 T magnetic resonance images in patients with HFS and control subjects; age, sex and hypertension were matched...
August 20, 2016: Parkinsonism & related Disorders
Jose Gutierrez, Khaled Menshawy, James Goldman, Andrew J Dwork, Mitchell S V Elkind, Randolph S Marshall, Susan Morgello
BACKGROUND:  To test the hypothesis that increased elastolytic activity would be associated differentially with dolichoectasia in individuals with and without HIV. METHODS:  Brain large arteries were obtained from 84 HIV+ and 78 HIV- autopsies and stained for metalloproteinase (MMP)-2, MMP-3, MMP-9, tissue inhibitor of metalloproteinases (TIMP)-1, TIMP-2, CD68, and caspase-3. Average Pixel Intensity was automatically obtained and categorized as high, moderate or low...
August 22, 2016: Journal of Infectious Diseases
Ignacio Arrese, Rosario Sarabia
INTRODUCTION: Vertebrobasilar dolichoectasia is a condition in which there is elongation and dilatation of the vertebral and basilar arteries. Few studies have been reported that focus on cases of trigeminal neuralgia (TN) secondary to vertebrobasilar dolichoectasia (VD) and treated by microvascular decompression (MD). PATIENTS AND METHODS: A case is presented of trigeminal neuralgia caused by vertebral artery compression. An analysis of the microsurgical technique, as well as a systematic review of the literature about this uncommon nerve compression is performed, in order to investigate, by pooled case analysis, if MD is a good option for this type of patient...
July 18, 2016: Neurocirugía
Arseny A Sokolov, Shakir Husain, Roman Sztajzel, Alexandre Croquelois, Johannes A Lobrinus, David Thaler, Claudio Städler, Hansjörg Hungerbühler, Valeria Caso, Gabriel J Rinkel, Patrik Michel
Vertebrobasilar dolichoectasia (VBD) is a chronic disorder with various cerebrovascular and compressive manifestations, involving subarachnoid hemorrhage (SAH). Occurrence of SAH shortly after worsening of clinical VBD symptoms has occasionally been reported. The goal of the study was to examine this association, in particular its pathophysiology, clinical precursor signs, time course, and outcome.To this end, in a retrospective multicenter study, we analyzed 20 patients with VBD and SAH in regard to preceding clinical symptoms, presence of vertebrobasilar thrombosis and ischemia, outcome and neuropathological correlates...
July 2016: Medicine (Baltimore)
Yue-Qi Zhu, Da-Ying Dai, Hai-Xia Xing, Yong-Hong Ding, Ramanathan Kadirvel, David F Kallmes
OBJECTIVE: To assess the feasibility of using MicroFil polymer perfusion to detect concomitant saccular aneurysms in an intracranial arterial dolichoectasia (IADE) model in mice, and to report detailed histomorphometric features of these aneurysms. MATERIALS AND METHODS: IADE models were created in C57/BL6 mice via microsurgical injection of 25 mU elastase into the cisterna magna. The cerebral vasculature was perfused with MicroFil polymer and harvested at 1, 3, and 7 days, and 2 and 4 weeks (n=8 for each group)...
July 5, 2016: Journal of Neurointerventional Surgery
Mohammad Samim, Alan Goldstein, Joseph Schindler, Michele H Johnson
Vertebrobasilar dolichoectasia (VBD) is characterized by ectasia, elongation, and tortuosity of the vertebrobasilar arteries, with a high degree of variability in clinical presentation. The disease origin is believed to involve degeneration of the internal elastic lamina, thinning of the media secondary to reticular fiber deficiency, and smooth muscle atrophy. The prevalence of VBD is variable, ranging from 0.05% to 18%. Most patients with VBD are asymptomatic and their VBD is detected incidentally; however, it is important to recognize that the presence of symptoms, which can lead to clinically significant morbidity and sometimes mortality, may influence clinical management...
July 2016: Radiographics: a Review Publication of the Radiological Society of North America, Inc
Xi Wu, Yanjun Li, Qinghai Huang, Jianmin Liu
BACKGROUND Vertebrobasilar dolichoectasia (VBD) is characterized by abnormal dilation, distortion, and extension of the vertebral artery (VA) and basilar artery (BA). This study investigated whether BA and VA morphological characteristics were factors predicting VBD. MATERIAL AND METHODS Individuals aged ≥18 years undergoing contrast-enhanced magnetic resonance angiography (CE-MRA) of the head/neck were enrolled in 2012 at Changhai Hospital, Shanghai. Data concerning cardiovascular risk factors were recorded...
2016: Medical Science Monitor: International Medical Journal of Experimental and Clinical Research
Mohammad Reza Najafi, Nafiseh Toghianifar, Morteza Abdar Esfahani, Mohammad Amin Najafi, Mohammad Javad Mollakouchakian
BACKGROUND: Vertebrobasilar dolichoectasia (VBD) is a rare vasculopathy. The etiology of this disease is unknown. Transient ischemic attacks (TIAs) of vertebrobasilar system refer to a transient (< 24 hours) lowering of blood flow in the posterior circulation of the brain. We present a case of dolichoectasia in the vertebrobasilar artery that presented with TIAs. CASE REPORT: A hypertensive 54-year-old man with true vertigo, nausea, imbalance, dysarthria, dysmetria, horizontal nystagmus, and gait ataxia was referred to Alzahra Hospital, Isfahan, Iran...
January 2016: ARYA Atherosclerosis
G Serebrinsky, M Calvo, S Fernandez, S Saito, K Ohno, E Wallace, D Warnock, H Sakuraba, J Politei
BACKGROUND: Screening for Fabry disease (FD) in high risk populations yields a significant number of individuals with novel, ultra rare genetic variants in the GLA gene, largely without classic manifestations of FD. These variants often have significant residual α-galactosidase A activity. The establishment of the pathogenic character of previously unknown or rare variants is challenging but necessary to guide therapeutic decisions. OBJECTIVES: To present 2 cases of non-classical presentations of FD with renal involvement as well as to discuss the importance of high risk population screenings for FD...
September 2015: Molecular Genetics and Metabolism Reports
Vikram Huded, Vikram Bohra, Jignesh Prajapati, Romnesh DeSouza, Ritesh Ramankutty
IMPORTANCE: In almost a third of stroke in young cases, etiology remains unclear. We report a radiological entity which might give a clue toward detection of late onset Pompe's disease in some cases. OBSERVATION: Here we report two cases of stroke in young in which evaluation led to diagnosis of late onset Pompe's disease. Both patients presented with recurrent stroke. On evaluation, one of them was found to have slowly progressive predominantly proximal quadriparesis...
April 2016: Journal of Stroke and Cerebrovascular Diseases: the Official Journal of National Stroke Association
Federica Montagnese, Francesca Granata, Olimpia Musumeci, Carmelo Rodolico, Stefania Mondello, Emanuele Barca, Maria Cucinotta, Anna Ciranni, Marcello Longo, Antonio Toscano
BACKGROUND AND OBJECTIVES: Pompe disease is a rare metabolic disorder due to lysosomal alpha-glucosidase (GAA) deficiency. It is considered as a multi-systemic disease since, although glycogen accumulation is largely prominent in heart, skeletal and respiratory muscles, other organs can also be affected. As regards the vascular system, few reports have documented cerebrovascular malformations in Pompe patients. The aim of this study was to define the presence and type of intracranial arterial abnormalities in a cohort of late onset Pompe disease (LOPD) patients...
May 2016: Journal of Inherited Metabolic Disease
Jose Gutierrez, Khaled Menshawy, Marco Gonzalez, James Goldman, Mitchell S V Elkind, Randolph Marshall, Susan Morgello
OBJECTIVE: To test the hypothesis that brain arteries from HIV+ cases have a greater degree of inflammation than brain arteries from HIV- cases, and that inflammation is associated with brain arterial remodeling. DESIGN: Case-control study, cross-sectional. METHODS: Brain arteries from 162 autopsy cases (84 with HIV) were systematically analyzed for thickness of the intima, media, and adventitia, and atherosclerosis and dolichoectasia. Inflammation was assessed with CD68 immunohistochemistry, and measured with a semiquantitative score reflecting the number and location (i...
January 28, 2016: AIDS
Hidetoshi Matsukawa, Masaki Shinoda, Motoharu Fujii, Akihiro Uemura, Osamu Takahashi, Yasunari Niimi
OBJECT: Basilar dolichoectasia (BD) is an atherosclerotic, a distinct arteriopathy or a chronic-phase dissection characterized by elongation and dilation of the basilar artery. Spontaneous intradural vertebral artery dissection (siVAD) is an important cause of stroke in young and middle-aged people. It is hypothesized that the BD and the siVAD might partially share aetiologies and this study aimed to examine the relationship. METHODS: This study compared clinical and radiological characteristics in 93 patients with siVAD with 93 controls...
2016: Brain Injury: [BI]
Mustafa AbdelHamid, Kuruvilla John, Tanvir Rizvi, Nicholas Huff
Hemifacial spasm (HFS) happens because of vascular compression of the facial nerve at the root exit zone. Vertebrobasilar dolichoectasia (VBD) is a very rare cause of HFS. VBD is diagnosed by computed tomography angiography and magnetic resonance imaging. Here, we report a case of 65-year-old female patient with HFS due to VBD. We discuss the complications and the treatment options for the case.
December 2015: Radiology case reports
Jose Gutierrez, James Goldman, Andrew J Dwork, Mitchell S V Elkind, Randolph S Marshall, Susan Morgello
BACKGROUND: Cerebrovascular disease is a cause of morbidity in HIV-infected populations. The relationship among HIV infection, brain arterial remodeling, and stroke is unclear. METHODS: Large brain arteries (n = 1,878 segments) from 284 brain donors with and without HIV were analyzed to obtain media and wall thickness and lumen-to-wall ratio, and to determine the presence of atherosclerosis and dolichoectasia (arterial remodeling extremes). Neuropathologic assessment was used to characterize brain infarcts...
September 29, 2015: Neurology
Ronald D Perrone, Adel M Malek, Terry Watnick
Autosomal dominant polycystic kidney disease (ADPKD) is the most common hereditary kidney disease. Relentless cyst growth substantially enlarges both kidneys and culminates in renal failure. Patients with ADPKD also have vascular abnormalities; intracranial aneurysms (IAs) are found in ∼10% of asymptomatic patients during screening and in up to 25% of those with a family history of IA or subarachnoid haemorrhage. As the genes responsible for ADPKD—PKD1 and PKD2—have complex integrative roles in mechanotransduction and intracellular calcium signalling, the molecular basis of IA formation might involve focal haemodynamic conditions exacerbated by hypertension and altered flow sensing...
October 2015: Nature Reviews. Nephrology
Vaso Zisimopoulou, Aikaterini Ntouniadaki, Panagiotis Aggelidakis, Anna Siatouni, Stylianos Gatzonis, Antonios Tavernarakis
Vertebrobasilar dolichoectasia is a clinical entity associated rarely with obstructive hydrocephalus. We present a 48-year old male with a profound dilatation of the ventricular system due to a dolichoectatic basilar artery, as appeared in imaging studies. The patient suffered from longstanding hydrocephalus and presenile dementia. The underlying mechanism for obstructive hydrocephalus due to vertebrobasilar dolichoectasia is considered to be both a water-hammer effect and a direct compression of adjacent structures...
April 24, 2015: Clinics and Practice
Fernando Pico, Julien Labreuche, Pierre Amarenco
Up to 12% of patients with stroke have intracranial arterial dolichoectasia (IADE) and the basilar artery is affected in 80% of these cases. Diagnostic criteria and prognosis studies of IADE are based on basilar artery diameter, which is a good quantitative marker for the severity of the disease. The pathophysiology is largely unknown, but IADE can be viewed as a common final pathway of arterial wall response or damage in the tunica media due to various mechanisms, such as matrix metalloproteinase dysfunction or muscle cell or elastic fibre injury...
August 2015: Lancet Neurology
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