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The Pipeline Embolization Device (PED) is a flow-diverting intraluminal device that is approved for use in adults 18 years or older with internal carotid artery aneurysms. However, it can also be used off-label in pediatric patients with aneurysms that cannot be resolved with traditional endovascular treatments. Herein, we present two cases of flow diversion in the pediatric population with complete obliteration of the aneurysm and excellent outcomes. Flow diversion has been shown to be a safe endovascular option in treating complex aneurysms in children. Larger-sized, multicenter trials are encouraged to compare outcomes between flow diversion and other aneurysm treatment options given the rarity of pediatric aneurysms.
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The development of pseudoaneurysm is an uncommon, life-threatening complication of head and neck microvascular surgery. Only a handful of reports have been published describing microvascular pseudoaneurysms, which usually occur at the arterial anastomosis and present as a pulsatile neck mass or as haemorrhage in case of pseudoaneurysm rupture. Management is highly variable, especially in the acute setting. In patients with pseudoaneurysm where flap inosculation is inadequate, endovascular approaches may be appropriate. In this report, we describe a ruptured distal pedicle pseudoaneurysm of a radial forearm free flap salvaged with a flow-diverting stent with complete flap survival and pedicle preservation. We demonstrate further evidence and feasibility of endovascular treatment of a non-anastomotic pseudoaneurysm arising from small vessels when parent vascular integrity is critical to flap survival.
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Falso Aneurisma , Procedimentos Endovasculares , Retalhos de Tecido Biológico , Procedimentos de Cirurgia Plástica , Humanos , Retalhos de Tecido Biológico/irrigação sanguínea , Falso Aneurisma/diagnóstico por imagem , Falso Aneurisma/etiologia , Falso Aneurisma/cirurgia , Artéria Radial/cirurgia , Procedimentos de Cirurgia Plástica/efeitos adversos , Procedimentos Endovasculares/efeitos adversosRESUMO
BACKGROUND AND OBJECTIVES: Complementary metal oxide semiconductor (CMOS) electrode arrays are a novel technology for miniaturized endoscopes; however, its use for neurointervention is yet to be investigated. In this proof-of-concept study, we aimed to demonstrate the feasibility of CMOS endoscopes in a canine model by providing direct visualization of the endothelial surface, deploying stents and coils, and accessing the spinal subdural space and skull base. METHODS: Using 3 canine models, standard guide catheters were introduced into the internal carotid and vertebral arteries through the transfemoral route using fluoroscopy. A 1.2-mm CMOS camera was delivered through the guide catheter to inspect the endothelium. Next, the camera was introduced alongside standard neuroendovascular devices including coils and stents to provide direct visualization of their deployment within the endothelium during fluoroscopy. One canine was used for skull base and extravascular visualization. A lumbar laminectomy was performed, and the camera was navigated within the spinal subdural space until the posterior circulation intracranial vasculature was visualized. RESULTS: We successfully visualized the endothelial surface and performed several endovascular procedures such as deployment of coils and stents under direct endovascular, angioscopic vision. We also demonstrated a proof of concept for accessing the skull base and posterior cerebral vasculature using CMOS cameras through the spinal subdural space. CONCLUSION: This proof-of-concept study demonstrates the feasibility of CMOS camera technology to directly visualize endothelium, perform common neuroendovascular procedures, and access the base of the skull in a canine model.
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Procedimentos Endovasculares , Base do Crânio , Animais , Cães , Base do Crânio/diagnóstico por imagem , Base do Crânio/cirurgia , Procedimentos Endovasculares/métodos , Stents , Fluoroscopia , CatéteresRESUMO
Background: Primary central nervous system lymphoma (PCNSL) is an aggressive extranodal subtype of nonHodgkin's lymphoma. Ventricle-predominant PCNSL, arising in the CNS ventricular system, is a rare entity. In over 90% of cases, PCNSL is classified as diffuse large B-cell lymphoma. Rarely, PCNSL may be classified as marginal zone B-cell lymphoma (MZBCL) of mucosa-associated lymphoid tissue (MALT). Taken together, a primary MALT-type MZBCL arising in a cerebral ventricle is an extremely rare presentation. Case Description: A 69-year-old female presented with a persistent left frontal headache for 1 year. Magnetic resonance imaging revealed an enhancing soft-tissue lesion within the left lateral ventricle, with associated periventricular edema. We performed an excisional biopsy of the tumor, which grossly had the appearance of a meningioma. Histopathology of the tumor was consistent with MZBCL of the MALT type. The patient was treated with Rituximab and Ibrutinib. Six months after surgery, she remained neurologically intact and free of disease. Conclusion: We report the case of a primary MALT-type MZBCL arising in the CNS ventricular system, with characteristics mimicking meningioma. This lymphoma involved the lateral ventricle and likely originated from the choroid plexus. Meningothelial cells and epithelial cells in the choroid plexus may acquire MALT in response to chronic inflammatory stimuli, such as infection or autoimmune disease. In rare cases, MALT lymphoma may develop as part of this pathogenesis.
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OBJECTIVE: Cervical artery dissection (CAD) has been associated with spinal manipulative therapy (SMT). Although uncommonly reported, SMT-associated CADs hold devastating neurological consequences, warranting further exploration. We endeavored to investigate this association through the comparison of all CAD etiologies at a single academic medical center. METHODS: A retrospective chart review was conducted of patients diagnosed with CAD or transferred to our institution for primary management of CAD during the 10-year period from 2010 to 2020 (n = 578). Patients were divided into SMT-associated (within 1 month of presentation), spontaneous, traumatic, and iatrogenic cohorts. RESULTS: SMT-associated dissections represented 23/578 (4%) of all dissections and 5.9% of vertebral artery dissections specifically. These patients were generally younger than those in the spontaneous (p = .004) and iatrogenic groups (p < .001), and more often non-smokers or former smokers compared to the spontaneous (p = .009), traumatic (p = .001), and iatrogenic (p = .008) groups. Additionally, the SMT group had a higher mean low-density lipoprotein (LDL) than the spontaneous (p = .009) and traumatic (p = .003) types. SMT-associated CADs were more often vertebral and bilateral, compared to the spontaneous (p = .003; p < .001), traumatic (p = .047; p = .004), and iatrogenic (p = .002; p = .002) groups. Outcomes including infarct (p = .112), medical treatment (p = .523), intervention (p = .47), and length of stay (p = .512) were similar between the SMT and spontaneous groups. CONCLUSIONS: In this unique study comparing SMT-associated CADs with other dissection etiologies, SMT-associated CADs were uncommon and not associated with worse clinical outcomes. However, SMT-associated CADs were more likely to be bilateral and affected the vertebral arteries in young, non-smoking patients with high LDL.
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Dissecação da Artéria Carótida Interna/etiologia , Manipulação da Coluna/efeitos adversos , Dissecação da Artéria Vertebral/etiologia , Centros Médicos Acadêmicos , Adulto , Idoso , Feminino , Humanos , Masculino , Pessoa de Meia-Idade , Estudos RetrospectivosRESUMO
Anesthesia for intracranial vascular procedures is complex because it requires a balance of several competing interests and potentially can result in significant morbidity and mortality. Frequently, periods of ischemia, where perfusion must be maintained, are combined with situations that are high risk for hemorrhage. This review discusses the basic surgical approach to several common pathologies (intracranial aneurysms, arteriovenous malformations, and moyamoya disease) along with the goals for anesthetic management and specific high-yield recommendations.
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Aneurisma Intracraniano , Doença de Moyamoya , Hemorragia Subaracnóidea , Humanos , Aneurisma Intracraniano/cirurgia , Doença de Moyamoya/cirurgiaRESUMO
Spinal dural arteriovenous fistula (SDAVF) is a rare pathological communication between arterial and venous vessels within the spinal dural sheath. Clinical presentation includes progressive spinal cord symptoms including gait difficulty, sensory disturbances, changes in bowel or bladder function, and sexual dysfunction. These fistulas are most often present in the thoracolumbar region. Diagnoses of SDVAFs are commonly missed, possibly due to the low index of suspicion, non-specific symptoms and challenging imaging. In this case report, we describe a rare presentation of a sacral SDAVF which was detected by collective efforts between endovascular neurosurgery and interventional radiology. We outline the diagnostic and imaging challenges we faced to discover the fistula. In particular, mechanical pump injection instead of hand injection during angiography was required to reveal the fistula. Following identification, the fistula was successfully treated endovascularly by using onyx (ethylene vinyl alcohol glue), a less invasive alternative to surgical intervention.
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Malformações Vasculares do Sistema Nervoso Central/diagnóstico por imagem , Malformações Vasculares do Sistema Nervoso Central/terapia , Embolização Terapêutica/métodos , Sacro/irrigação sanguínea , Angiografia , Diagnóstico Diferencial , Humanos , Imageamento por Ressonância Magnética , Masculino , Pessoa de Meia-IdadeRESUMO
PURPOSE: Variations in the vessel radius of segmented surfaces of intracranial aneurysms significantly influence the fluid velocities given by computer simulations. It is important to generate models that capture the effect of these variations in order to have a better interpretation of the numerically predicted hemodynamics. Also, it is highly relevant to develop methods that combine experimental observations with uncertainty modeling to get a closer approximation to the blood flow behavior. METHODS: This work applies polynomial chaos expansion to model the effect of geometric uncertainties on the simulated fluid velocities of intracranial aneurysms. The radius of the vessel is defined as the uncertainty variable. Proper orthogonal decomposition is applied to characterize the solution space of fluid velocities. Next, a process of projecting the 4D-Flow MRI velocities on the basis vectors followed by coefficient mapping using generalized dynamic mode decomposition enables the merging of 4D-Flow MRI with the uncertainty propagated fluid velocities. RESULTS: Polynomial chaos expansion propagates the fluid velocities with an error of 2% in velocity magnitude relative to computer simulations. Also, the bifurcation region (or impingement location) shows a standard deviation of 0.17 m/s (since an available reported variance in the vessel radius is adopted to model the uncertainty, the expected standard deviation may be different). Numerical phantom experiments indicate that the proposed approach reconstructs the fluid velocities with 0.3% relative error in presence of geometric uncertainties. CONCLUSION: Polynomial chaos expansion is an effective approach to propagate the effect of the uncertainty variable in the blood flow velocities of intracranial aneurysms. Merging 4D-Flow MRI and uncertainty propagated fluid velocities leads to more realistic flow trends relative to ignoring the uncertainty in the vessel radius.
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Aneurisma Intracraniano , Velocidade do Fluxo Sanguíneo , Hemodinâmica , Humanos , Aneurisma Intracraniano/diagnóstico por imagem , Imageamento por Ressonância Magnética , IncertezaRESUMO
4D-Flow magnetic resonance imaging (MRI) has enabled in vivo time-resolved measurement of three-dimensional blood flow velocities in the human vascular system. However, its clinical use has been hampered by two main issues, namely, low spatio-temporal resolution and acquisition noise. While patient-specific computational fluid dynamics (CFD) simulations can address the resolution and noise issues, its fidelity is impacted by accuracy of estimation of boundary conditions, model parameters, vascular geometry, and flow model assumptions. In this paper a scheme to address limitations of both modalities through data-fusion is presented. The solutions of the patient-specific CFD simulation are characterized using proper orthogonal decomposition (POD). Next, a process of projecting the 4D-Flow MRI data onto the POD basis and projection coefficient mapping using generalized dynamic mode decomposition (DMD) enables simultaneous super-resolution and denoising of 4D-Flow MRI. The method has been tested using numerical phantoms derived from patient-specific aneurysmal geometries and applied to in vivo 4D-Flow MRI data.
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Hidrodinâmica , Imageamento por Ressonância Magnética , Velocidade do Fluxo Sanguíneo , Humanos , Imageamento Tridimensional , Imagens de FantasmasRESUMO
4D-Flow MRI has emerged as a powerful tool to non-invasively image blood velocity profiles in the human cardio-vascular system. However, it is plagued by issues such as velocity aliasing, phase offsets, acquisition noise, and low spatial and temporal resolution. In imaging small blood vessel malformations such as intra-cranial aneurysms, the spatial resolution of 4D-Flow is often inadequate to resolve fine flow features. In this paper, we address the problem of low spatial resolution and noise by combining 4D-Flow MRI and patient specific computational fluid dynamics using Least Absolute Shrinkage and Selection Operator. Extensive experiments using numerical phantoms of two actual intra-cranial aneurysms geometries show the applicability of the proposed method in recovering the flow profile. Comparisons with the state-of-the-art denoising methods for 4D-Flow show lower error metrics. This method can enable more accurate computation of flow derived patho-physiological parameters such as wall shear stresses, pressure gradients, and viscous dissipation.
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Hidrodinâmica , Imageamento Tridimensional/métodos , Aneurisma Intracraniano/diagnóstico por imagem , Imageamento por Ressonância Magnética/métodos , Imagens de Fantasmas , Razão Sinal-Ruído , Algoritmos , Velocidade do Fluxo Sanguíneo , HumanosRESUMO
Superficial temporal arterial to middle cerebral arterial anastomosis is often the initial surgical treatment of Moyamoya disease. In refractory cases, placing a pedicle flap of omentum over the ischemic brain has resulted in clinical improvement or stabilization of symptoms. We present a case of persistent mesenteric traction syndrome manifested by hypotension unresponsive to conventional doses of vasopressors during and after pulling the omentum to the brain. As prostacyclin is a major mediator of hypotension from mesenteric traction syndrome and also a cerebral vasodilator, we discuss the possibility that brain swelling may be a manifestation of mesenteric traction syndrome.
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Edema Encefálico/etiologia , Hipotensão/etiologia , Doença de Moyamoya/cirurgia , Retalhos Cirúrgicos/efeitos adversos , Gerenciamento Clínico , Feminino , Humanos , Omento/cirurgia , Adulto JovemRESUMO
Crossing the neck of large complex intracranial aneurysms for the purposes of stent deployment can be challenging using standard over the wire techniques. We describe a novel yet simple technique for straightening out the loop formed within a large intracranial aneurysm, which is often required in order to cross the aneurysm neck into the distal branch. Both the microcatheter and microwire are initially introduced into the distal vasculature, followed by withdrawal of the microwire to a point parallel to the distal exiting branch. The microcatheter and microwire are then gently withdrawn and a series of maneuvers to gradually reduce the loop is performed, obviating the need for distal purchase in the form of a stent, balloon, or coil, which have previously been described to maintain distal purchase.
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Procedimentos Endovasculares/métodos , Aneurisma Intracraniano/diagnóstico por imagem , Aneurisma Intracraniano/cirurgia , Stents , Idoso , Cateterismo/instrumentação , Cateterismo/métodos , Procedimentos Endovasculares/instrumentação , Feminino , HumanosRESUMO
BACKGROUND: Open surgery is a frequent option given to patients with unruptured intracranial aneurysms (UIAs) unsuitable for endovascular repair. Since the risk of rupture of UIAs is generally low, we determined whether the risks and costs of surgery in this patient subset are warranted. METHODS: The safety, efficacy, and costs of minimally invasive surgery by minicraniotomy were evaluated in 102 consecutive patients with anterior circulation UIAs deemed unsuitable for endovascular repair by an interdisciplinary conference of surgeons and neurointerventionalists. Data from 107 UIA patients treated by endovascular means in the same period were used as the standard. RESULTS: Surgical patients comprised a different subset of aneurysms, with more MCA and fewer paraophthalmic aneurysms (54 vs. 6, p < 0.0001 and 4 vs. 60, p < 0.0001, for minicraniotomy and endovascular, respectively). However, surgery incurred shorter anesthesia time (197.7 vs. 149.3 min, p < 0.0001), higher rates of complete aneurysm obliteration (94.57 vs. 66.67 %, p < 0.0001), and lower overall hospital costs ($8,287 CAD vs. $17,732 CAD, p < 0.0001) than the endovascular cohort. There were no treatment-related surgical deaths, but one patient had an mRS of 3 after 6 months due to temporal lobe epilepsy and memory problems. This compared favorably with the endovascular cohort in which two patients died due to treatment (mRS = 6) and one suffered a severe stroke (mRS = 5 at 6 months). CONCLUSIONS: For patients counseled to undergo treatment but have UIAs unsuitable for endovascular repair, surgery is safe, effective, and cost-efficient.
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Embolização Terapêutica/efeitos adversos , Aneurisma Intracraniano/cirurgia , Procedimentos Cirúrgicos Minimamente Invasivos/efeitos adversos , Custos e Análise de Custo , Embolização Terapêutica/economia , Embolização Terapêutica/métodos , Feminino , Humanos , Masculino , Pessoa de Meia-Idade , Procedimentos Cirúrgicos Minimamente Invasivos/economia , Procedimentos Cirúrgicos Minimamente Invasivos/métodosRESUMO
BACKGROUND AND PURPOSE: Basilar trunk aneurysms (BTAs), defined as aneurysms distal to the basilar origin and proximal to the origin of the superior cerebellar artery, are rare and challenging to manage. We describe the natural history and management in a consecutive series of BTAs. METHODS: Between 2000 and 2013, 2522 patients with 3238 aneurysms were referred to our institution for aneurysm management. A retrospective review of this database was conducted to identify all patients with BTAs. RESULTS: In total, 52 patients had a BTA. Mean age was 56 (SD±18) years. Median clinical follow-up was 33 (interquartile range, 8-86) months, and imaging follow-up was 26 (interquartile range, 2-80.5) months. BTAs were classified into 4 causal subtypes: acute dissecting aneurysms, segmental fusiform ectasia, mural bleeding ectasia, and saccular aneurysms. Multiple aneurysms were more frequently noticed among the 13 saccular aneurysms when compared with overall population (P=0.021). There was preponderance of segmental ectasia or mural bleeding ectasia (P=0.045) in patients presenting with transit ischemic attack/stroke or mass effect. Six patients with segmental and 4 with mural bleeding ectasia demonstrated increasing size of their aneurysm, with 2 having subarachnoid hemorrhage caused by aneurysm rupture. None of the fusiform aneurysms that remained stable bled. CONCLUSIONS: BTAs natural histories may differ depending on subtype of aneurysm. Saccular aneurysms likely represent an underlying predisposition to aneurysm development because more than half of these cases were associated with multiple intracranial aneurysms. Intervention should be considered in segmental ectasia and chronic dissecting aneurysms, which demonstrate increase in size over time as there is an increased risk of subarachnoid hemorrhage.
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Artéria Basilar/patologia , Aneurisma Intracraniano/classificação , Adulto , Idoso , Idoso de 80 Anos ou mais , Dissecção Aórtica/patologia , Dissecção Aórtica/terapia , Dilatação Patológica/patologia , Dilatação Patológica/terapia , Progressão da Doença , Suscetibilidade a Doenças , Feminino , Humanos , Aneurisma Intracraniano/patologia , Aneurisma Intracraniano/terapia , Masculino , Pessoa de Meia-Idade , Estudos RetrospectivosRESUMO
Crossing the neck of large complex intracranial aneurysms for the purposes of stent deployment can be challenging using standard over the wire techniques. We describe a novel yet simple technique for straightening out the loop formed within a large intracranial aneurysm, which is often required in order to cross the aneurysm neck into the distal branch. Both the microcatheter and microwire are initially introduced into the distal vasculature, followed by withdrawal of the microwire to a point parallel to the distal exiting branch. The microcatheter and microwire are then gently withdrawn and a series of maneuvers to gradually reduce the loop is performed, obviating the need for distal purchase in the form of a stent, balloon, or coil, which have previously been described to maintain distal purchase.
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Catéteres , Embolização Terapêutica/métodos , Aneurisma Intracraniano/terapia , Stents , Idoso , Feminino , Humanos , TraçãoRESUMO
Cerebral revascularization is used to augment or replace cerebral blood flow in patients at risk of developing cerebral ischemia. These include patients with moyamoya disease, occlusive cerebrovascular disease, skull base tumors, and complex aneurysms. Our aim in this review is to provide a comprehensive update of both surgical and anesthetic aspects of cerebral revascularization procedures. The anesthetic concerns for most patients presenting for different types of bypass procedures are similar and include the maintenance of adequate cerebral perfusion to prevent cerebral ischemia. Patients with complex aneurysms and tumors have additional considerations related to the surgical treatment of the underlying pathology.
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Anestesia , Revascularização Cerebral/métodos , Revascularização Cerebral/efeitos adversos , Circulação Cerebrovascular/fisiologia , Transtornos Cerebrovasculares/cirurgia , Humanos , Complicações Pós-Operatórias/terapiaRESUMO
BACKGROUND AND PURPOSE: Management of unruptured fusiform intracranial aneurysms is controversial because of the paucity of natural history data. We studied their natural history and outcome after treatment. METHODS: We reviewed our neurovascular database from January 2000 to October 2013. Inclusion criteria were unruptured, intradural fusiform aneurysms with a diameter of <2.5 cm. Criteria were developed to define atherosclerotic aneurysms. For outcome assessment, we used the modified Ranking Scale and aneurysm measurements on serial imaging. Mann-Whittney (continuous) and Fisher exact (categorical) tests were used for risk factor analysis. RESULTS: For nonatherosclerotic aneurysms (96 patients; 193 person-years follow-up), 1 patient died (rupture) during follow-up (mortality, 0.51% per year) and 8 patients (10%) showed aneurysm progression (risk, 1.6% per year). Risk factors for progression were maximum diameter (>7 mm; odds ratio, 12; 95% confidence interval, 1.4-104) and symptomatic clinical presentation (odds ratio, 16; 95% confidence interval, 3.1-81.4). Of the 23 treated patients, 3 had died (mortality, 12.5%) and 3 had serious disability (modified Ranking Scale, ≥3; 12.5%). For the atherosclerotic aneurysms (25 patients; 97 person-years follow-up), 5 had died (mortality, 5.2% per year) and 13 of 20 (65%) had aneurysm progression (risk, 12% per year). When compared with patients with nonatherosclerotic aneurysms, case fatality (odds ratio, 19.2; 95% confidence interval, 2.1-172) and aneurysm progression (odds ratio, 17.8; 95% confidence interval, 5.3-56) were higher. CONCLUSIONS: Nonatherosclerotic fusiform intradural aneurysms have a low risk of adverse outcome within the first few years after diagnosis and remain stable unless symptomatic on presentation or >7 mm in maximum diameter. High risks of treatment should be balanced against this benign natural history. Atherosclerotic aneurysms have a worse natural history and may represent a different disease entity.
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Bases de Dados Factuais , Aneurisma Intracraniano/epidemiologia , Aneurisma Intracraniano/terapia , Adolescente , Adulto , Idoso , Idoso de 80 Anos ou mais , Criança , Bases de Dados Factuais/tendências , Feminino , Seguimentos , Humanos , Aneurisma Intracraniano/diagnóstico , Masculino , Pessoa de Meia-Idade , Estudos Prospectivos , Estudos Retrospectivos , Fatores de Risco , Resultado do Tratamento , Adulto JovemRESUMO
BACKGROUND: Endoluminal optical imaging, or angioscopy, has not seen widespread application during neurointerventional procedures, largely as a result of the poor imaging resolution of existing angioscopes. Scanning fiber endoscopes (SFEs) are a novel endoscopic platform that allows high-resolution video imaging in an ultraminiature form factor that is compatible with currently used distal access endoluminal catheters. OBJECTIVE: To test the feasibility and potential utility of high-resolution angioscopy with an SFE during common endovascular neurosurgical procedures. METHODS: A 3.7-French SFE was used in a porcine model system to image endothelial disruption, ischemic stroke and mechanical thrombectomy, aneurysm coiling, and flow-diverting stent placement. RESULTS: High-resolution, video-rate imaging was shown to be possible during all of the common procedures tested and provided information that was complementary to standard fluoroscopic imaging. SFE angioscopy was able to assess novel factors such as aneurysm base coverage fraction and side branch patency, which have previously not been possible to determine with conventional angiography. CONCLUSION: Endovascular imaging with an SFE provides important information on factors that cannot be assessed fluoroscopically and is a novel platform on which future neurointerventional techniques may be based because it allows for periprocedural inspection of the integrity of the vascular system and the deployed devices. In addition, it may be of diagnostic use for inspecting the vascular wall and postprocedure device evaluation.
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Angioscopia/instrumentação , Acidente Vascular Cerebral/diagnóstico , Acidente Vascular Cerebral/cirurgia , Animais , Modelos Animais de Doenças , Endoscopia , Estudos de Viabilidade , Procedimentos Neurocirúrgicos/métodos , SuínosRESUMO
The development of a traumatic arteriovenous fistula after arthroscopic surgery of the temporomandibular joint (TMJ) is an unusual event that has been limited to a few case reports. These have generally involved the superficial temporal artery and surrounding venous outlets. No cases of either postoperative or post-traumatic arteriovenous fistulas involving the extracranial middle meningeal artery (MMA), which is located on the medial surface of the TMJ, have been previously reported. We report, to our knowledge, the first case of this unusual complication and describe its successful endovascular management.
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Fístula Arteriovenosa/cirurgia , Artroscopia/métodos , Procedimentos Endovasculares , Artérias Meníngeas/anormalidades , Transtornos da Articulação Temporomandibular/cirurgia , Articulação Temporomandibular/cirurgia , Adulto , Angiografia , Feminino , Humanos , Artérias Meníngeas/cirurgiaRESUMO
STUDY DESIGN: A retrospective radiographical follow-up study of thoracolumbar deformity in 33 children with mucopolysaccharidosis 1 (Hurler syndrome). OBJECTIVE: To report the severity, natural history, risk factors for progression, and results of intervention for thoracolumbar kyphosis in children with Hurler syndrome. SUMMARY OF BACKGROUND DATA: Literature on the subject of thoracolumbar kyphosis in Hurler syndrome and its treatment is limited to small case series. The natural history and thus indications for intervention are unknown. METHODS: Patients who had been treated with bone marrow transplantation and/or enzyme replacement therapy were followed up with erect radiographs of the spine. Mean follow-up period was 3.5 years (range, 2-12 yr). Radiographs were retrieved and analyzed retrospectively. Seven patients underwent varied forms of surgical intervention for progressive deformity, the technique and principles of which are described. RESULTS: The thoracolumbar kyphosis on initial radiographs obtained at a mean age of 17 months measured 38° (95% confidence interval, 34°-42°). Fifteen of the 33 patients (45%) followed for more than 2 years developed a deformity that made a progression of more than 10°. The magnitude of the initial deformity was predictive of whether the deformity progressed (univariate analysis, P < 0.001). An initial kyphosis angle greater than 45° was predictive of progression of more than 10° with sensitivity of 67% and specificity of 88%. All patients who underwent surgical intervention had sustained improvement in the magnitude of thoracolumbar deformity. CONCLUSION: Thoracolumbar kyphosis in Hurler syndrome is of variable severity with an average deformity, in our series, of 38° at a mean age of 17 months. Forty-five percent of patients developed progression of greater than 10°. Patients with an initial deformity greater than 45° seemed to be more likely to progress. Surgical interventions in the form of anterior fusion, combined anterior and posterior surgery and use of the vertical expandable prosthetic titanium rib provided good correction. LEVEL OF EVIDENCE: N/A.