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1.
Nat Commun ; 13(1): 666, 2022 02 03.
Artigo em Inglês | MEDLINE | ID: mdl-35115552

RESUMO

Affecting 1.1‰ of infants, hydrocephalus involves abnormal accumulation of cerebrospinal fluid, resulting in elevated intracranial pressure (ICP). It is the leading cause for brain surgery in newborns, often causing long-term neurologic disabilities or even death. Since conventional invasive ICP monitoring is risky, early neurosurgical interventions could benefit from noninvasive techniques. Here we use clinical contrast-enhanced ultrasound (CEUS) imaging and intravascular microbubble tracking algorithms to map the cerebral blood flow in hydrocephalic pediatric porcine models. Regional microvascular perfusions are quantified by the cerebral microcirculation (CMC) parameter, which accounts for the concentration of micro-vessels and flow velocity in them. Combining CMC with hemodynamic parameters yields functional relationships between cortical micro-perfusion and ICP, with correlation coefficients exceeding 0.85. For cerebral ischemia cases, the nondimensionalized cortical micro-perfusion decreases by an order of magnitude when ICP exceeds 50% of the MAP. These findings suggest that CEUS-based CMC measurement is a plausible noninvasive method for assessing the ICP and detecting ischemia.


Assuntos
Circulação Cerebrovascular/fisiologia , Pressão Intracraniana/fisiologia , Isquemia/fisiopatologia , Microcirculação/fisiologia , Reologia/métodos , Animais , Isquemia Encefálica/diagnóstico , Isquemia Encefálica/diagnóstico por imagem , Isquemia Encefálica/fisiopatologia , Meios de Contraste , Eletrocardiografia , Feminino , Hemodinâmica/fisiologia , Humanos , Hidrocefalia/diagnóstico , Hidrocefalia/diagnóstico por imagem , Hidrocefalia/fisiopatologia , Lactente , Isquemia/diagnóstico , Isquemia/diagnóstico por imagem , Microbolhas , Reprodutibilidade dos Testes , Sensibilidade e Especificidade , Suínos , Ultrassonografia/métodos
2.
Biomolecules ; 11(8)2021 07 30.
Artigo em Inglês | MEDLINE | ID: mdl-34439789

RESUMO

We investigated cerebrospinal fluid (CSF) expression of inflammatory cytokines and their relationship with spontaneous intracerebral and intraventricular hemorrhage (ICH, IVH) and perihematomal edema (PHE) volumes in patients with acute IVH. Twenty-eight adults with IVH requiring external ventricular drainage for obstructive hydrocephalus had cerebrospinal fluid (CSF) collected for up to 10 days and had levels of interleukin-1α (IL-1α), IL-1ß, IL-6, IL-8, IL-10, tumor necrosis factor-α (TNFα), and C-C motif chemokine ligand CCL2 measured using enzyme-linked immunosorbent assay. Median [IQR] ICH and IVH volumes at baseline (T0) were 19.8 [5.8-48.8] and 14.3 [5.3-38] mL respectively. Mean levels of IL-1ß, IL-6, IL-10, TNF-α, and CCL2 peaked early compared to day 9-10 (p < 0.05) and decreased across subsequent time periods. Levels of IL-1ß, IL-6, IL-8, IL-10, and CCL2 had positive correlations with IVH volume at days 3-8 whereas positive correlations with ICH volume occurred earlier at day 1-2. Significant correlations were found with PHE volume for IL-6, IL-10 and CCL2 at day 1-2 and with relative PHE at days 7-8 or 9-10 for IL-1ß, IL-6, IL-8, and IL-10. Time trends of CSF cytokines support experimental data suggesting association of cerebral inflammatory responses with ICH/IVH severity. Pro-inflammatory markers are potential targets for injury reduction.


Assuntos
Hemorragia Cerebral Intraventricular/genética , Expressão Gênica , Hidrocefalia/genética , Adulto , Idoso , Hemorragia Cerebral Intraventricular/líquido cefalorraquidiano , Hemorragia Cerebral Intraventricular/fisiopatologia , Hemorragia Cerebral Intraventricular/terapia , Quimiocina CCL2/líquido cefalorraquidiano , Quimiocina CCL2/genética , Drenagem/métodos , Feminino , Humanos , Hidrocefalia/líquido cefalorraquidiano , Hidrocefalia/fisiopatologia , Hidrocefalia/terapia , Interleucina-10/líquido cefalorraquidiano , Interleucina-10/genética , Interleucina-1alfa/líquido cefalorraquidiano , Interleucina-1alfa/genética , Interleucina-1beta/líquido cefalorraquidiano , Interleucina-1beta/genética , Interleucina-6/líquido cefalorraquidiano , Interleucina-6/genética , Interleucina-8/líquido cefalorraquidiano , Interleucina-8/genética , Masculino , Pessoa de Meia-Idade , Estudos Prospectivos , Índice de Gravidade de Doença , Fator de Necrose Tumoral alfa/líquido cefalorraquidiano , Fator de Necrose Tumoral alfa/genética
3.
Fluids Barriers CNS ; 18(1): 31, 2021 Jul 07.
Artigo em Inglês | MEDLINE | ID: mdl-34233705

RESUMO

BACKGROUND: Cerebrospinal fluid (CSF) is an ultra-filtrated colorless brain fluid that circulates within brain spaces like the ventricular cavities, subarachnoid space, and the spine. Its continuous flow serves many primary functions, including nourishment, brain protection, and waste removal. MAIN BODY: The abnormal accumulation of CSF in brain cavities triggers severe hydrocephalus. Accumulating evidence had indicated that synchronized beats of motile cilia (cilia from multiciliated cells or the ependymal lining in brain ventricles) provide forceful pressure to generate and restrain CSF flow and maintain overall CSF circulation within brain spaces. In humans, the disorders caused by defective primary and/or motile cilia are generally referred to as ciliopathies. The key role of CSF circulation in brain development and its functioning has not been fully elucidated. CONCLUSIONS: In this review, we briefly discuss the underlying role of motile cilia in CSF circulation and hydrocephalus. We have reviewed cilia and ciliated cells in the brain and the existing evidence for the regulatory role of functional cilia in CSF circulation in the brain. We further discuss the findings obtained for defective cilia and their potential involvement in hydrocephalus. Furthermore, this review will reinforce the idea of motile cilia as master regulators of CSF movements, brain development, and neuronal diseases.


Assuntos
Encéfalo/fisiologia , Líquido Cefalorraquidiano/fisiologia , Cílios/fisiologia , Hidrocefalia/líquido cefalorraquidiano , Hidrocefalia/fisiopatologia , Animais , Encéfalo/citologia , Ventrículos Cerebrais/citologia , Ventrículos Cerebrais/fisiologia , Humanos
4.
Pediatr Clin North Am ; 68(4): 793-809, 2021 08.
Artigo em Inglês | MEDLINE | ID: mdl-34247710

RESUMO

Hydrocephalus is a pathologic condition that results in the disruption of normal cerebrospinal fluid flow dynamics often characterized by an increase in intracranial pressure resulting in an abnormal dilation of the ventricles. The goal of this article was to provide the necessary background information to understand the pathophysiology related to hydrocephalus, recognize the presenting signs and symptoms of hydrocephalus, identify when to initiate a workup with further studies, and understand the management of pediatric patients with a new and preexisting diagnosis of hydrocephalus.


Assuntos
Hidrocefalia/diagnóstico , Hidrocefalia/fisiopatologia , Atenção Primária à Saúde/organização & administração , Criança , Diagnóstico Diferencial , Humanos , Pressão Intracraniana/fisiologia , Monitorização Fisiológica , Fatores de Risco
5.
Fluids Barriers CNS ; 18(1): 25, 2021 May 26.
Artigo em Inglês | MEDLINE | ID: mdl-34039383

RESUMO

BACKGROUND: Idiopathic normal pressure hydrocephalus (iNPH) and late-onset idiopathic aqueductal stenosis (LIAS) are two forms of chronic adult hydrocephalus of different aetiology. We analysed overnight intracranial pressure (ICP) monitoring to elucidate ICP waveform changes characteristic for iNPH and LIAS to better understand pathophysiological processes of both diseases. METHODS: 98 patients with iNPH and 14 patients with LIAS from two neurosurgical centres were included. All patients underwent diagnostic overnight computerised ICP monitoring with calculation of mean ICP, ICP heartbeat related pulse wave amplitude calculated in the frequency domain (AMP) and the time domain (MWA), index of cerebrospinal compensatory reserve (RAP) and power of slow vasogenic waves (SLOW). RESULTS: ICP was higher in LIAS than iNPH patients (9.3 ± 3.0 mmHg versus 5.4 ± 4.2 mmHg, p = 0.001). AMP and MWA were higher in iNPH versus LIAS (2.36 ± 0.91 mmHg versus 1.81 ± 0.59 mmHg for AMP, p = 0.012; 6.0 ± 2.0 mmHg versus 4.9 ± 1.2 mmHg for MWA, p = 0.049). RAP and SLOW indicated impaired reserve capacity and compliance in both diseases, but did not differ between groups. INPH patients were older than LIAS patients (77 ± 6 years versus 54 ± 14 years, p < 0.001). CONCLUSIONS: ICP is higher in LIAS than in iNPH patients, likely due to the chronically obstructed CSF flow through the aqueduct, but still in a range considered normal. Interestingly, AMP/MWA was higher in iNPH patients, suggesting a possible role of high ICP pulse pressure amplitudes in iNPH pathophysiology. Cerebrospinal reserve capacity and intracranial compliance is impaired in both groups and the pressure-volume relationship might be shifted towards lower ICP values in iNPH. The physiological influence of age on ICP and AMP/MWA requires further research.


Assuntos
Pressão Sanguínea/fisiologia , Hidrocefalia de Pressão Normal/epidemiologia , Hidrocefalia de Pressão Normal/fisiopatologia , Hidrocefalia/epidemiologia , Hidrocefalia/fisiopatologia , Pressão Intracraniana/fisiologia , Adulto , Idade de Início , Idoso , Idoso de 80 Anos ou mais , Feminino , Humanos , Hidrocefalia/diagnóstico por imagem , Hidrocefalia de Pressão Normal/diagnóstico por imagem , Masculino , Pessoa de Meia-Idade
6.
J Pediatr ; 236: 54-61.e1, 2021 Sep.
Artigo em Inglês | MEDLINE | ID: mdl-34004191

RESUMO

OBJECTIVE: To demonstrate that a novel noninvasive index of intracranial pressure (ICP) derived from diffuse optics-based techniques is associated with intracranial hypertension. STUDY DESIGN: We compared noninvasive and invasive ICP measurements in infants with hydrocephalus. Infants born term and preterm were eligible for inclusion if clinically determined to require cerebrospinal fluid (CSF) diversion. Ventricular size was assessed preoperatively via ultrasound measurement of the fronto-occipital (FOR) and frontotemporal (FTHR) horn ratios. Invasive ICP was obtained at the time of surgical intervention with a manometer. Intracranial hypertension was defined as invasive ICP ≥15 mmHg. Diffuse optical measurements of cerebral perfusion, oxygen extraction, and noninvasive ICP were performed preoperatively, intraoperatively, and postoperatively. Optical and ultrasound measures were compared with invasive ICP measurements, and their change in values after CSF diversion were obtained. RESULTS: We included 39 infants, 23 with intracranial hypertension. No group difference in ventricular size was found by FOR (P = .93) or FTHR (P = .76). Infants with intracranial hypertension had significantly higher noninvasive ICP (P = .02) and oxygen extraction fraction (OEF) (P = .01) compared with infants without intracranial hypertension. Increased cerebral blood flow (P = .005) and improved OEF (P < .001) after CSF diversion were observed only in infants with intracranial hypertension. CONCLUSIONS: Noninvasive diffuse optical measures (including a noninvasive ICP index) were associated with intracranial hypertension. The findings suggest that impaired perfusion from intracranial hypertension was independent of ventricular size. Hemodynamic evidence of the benefits of CSF diversion was seen in infants with intracranial hypertension. Noninvasive optical techniques hold promise for aiding the assessment of CSF diversion timing.


Assuntos
Circulação Cerebrovascular/fisiologia , Hidrocefalia/diagnóstico por imagem , Hidrocefalia/fisiopatologia , Hipertensão Intracraniana/diagnóstico , Derivações do Líquido Cefalorraquidiano , Estudos de Viabilidade , Feminino , Humanos , Hidrocefalia/cirurgia , Recém-Nascido , Hipertensão Intracraniana/etiologia , Hipertensão Intracraniana/fisiopatologia , Pressão Intracraniana/fisiologia , Masculino , Imagem Óptica , Projetos Piloto , Reprodutibilidade dos Testes , Análise Espectral
7.
Fluids Barriers CNS ; 18(1): 20, 2021 Apr 19.
Artigo em Inglês | MEDLINE | ID: mdl-33874972

RESUMO

Idiopathic normal pressure hydrocephalus (iNPH) is considered an age-dependent chronic communicating hydrocephalus associated with cerebrospinal fluid (CSF) malabsorption; however, the aetiology of ventricular enlargement in iNPH has not yet been elucidated. There is accumulating evidence that support the hypothesis that various alterations in CSF dynamics contribute to ventricle dilatation in iNPH. This review focuses on CSF dynamics associated with ventriculomegaly and summarises the current literature based on three potential aetiology factors: genetic, environmental and hydrodynamic. The majority of gene mutations that cause communicating hydrocephalus were associated with an abnormal structure or dysfunction of motile cilia on the ventricular ependymal cells. Aging, alcohol consumption, sleep apnoea, diabetes and hypertension are candidates for the risk of developing iNPH, although there is no prospective cohort study to investigate the risk factors for iNPH. Alcohol intake may be associated with the dysfunction of ependymal cilia and sustained high CSF sugar concentration due to uncontrolled diabetes increases the fluid viscosity which in turn increases the shear stress on the ventricular wall surface. Sleep apnoea, diabetes and hypertension are known to be associated with the impairment of CSF and interstitial fluid exchange. Oscillatory shear stress to the ventricle wall surfaces is considerably increased by reciprocating bidirectional CSF movements in iNPH. Increased oscillatory shear stress impedes normal cilia beating, leading to motile cilia shedding from the ependymal cells. At the lack of ciliary protection, the ventricular wall is directly exposed to increased oscillatory shear stress. Additionally, increased oscillatory shear stress may be involved in activating the flow-mediated dilation signalling of the ventricular wall. In conclusion, as the CSF stroke volume at the cerebral aqueduct increases, the oscillatory shear stress increases, promoting motor cilia shedding and loss of ependymal cell coverage. These are considered to be the leading causes of ventricular enlargement in iNPH.


Assuntos
Líquido Cefalorraquidiano/fisiologia , Cílios/fisiologia , Epêndima/fisiopatologia , Hidrocefalia de Pressão Normal/fisiopatologia , Hidrocefalia/etiologia , Hidrocefalia/fisiopatologia , Humanos
9.
World Neurosurg ; 149: e651-e663, 2021 05.
Artigo em Inglês | MEDLINE | ID: mdl-33548530

RESUMO

BACKGROUND: Gangliogliomas (GGs) are extremely rare benign neoplasms frequently located within the temporal lobe that usually present with seizures. GGs growing predominantly within the ventricular system (VGGs) are even more infrequent, so definite conclusions concerning their diagnosis and therapeutic management are lacking. METHODS: A retrospective review of case reports of VGGs was performed from the introduction of modern imaging techniques, including 4 new illustrative cases treated in our department. RESULTS: Thirty-four cases were collected. Ages ranged from 10 to 71 years (mean, 26.62 years), and 55.9% were male. Most patients developed symptoms related to high intracranial pressure. The lateral ventricles were predominantly involved (58.8%). Obstructive hydrocephalus was observed in 54.5% of patients. Cystic degeneration and calcification were frequently observed. Surgical treatment was carried out in all cases. Morbidity and mortality were 17.6% and 2.9%, respectively. Gross total tumor resection was achieved in 64.5% of patients. Four patients experienced tumor dissemination along the neural axis. More than 90% of patients maintained a good functional status at last follow-up. CONCLUSIONS: Despite their low incidence, a diagnosis of VGGs should be considered in young male adults who progressively develop intracranial hypertension, caused by a ventricular mass showing signs of cystic degeneration and calcification. Maximal and safe surgical resection represents the gold standard for the treatment of symptomatic VGGs, although total removal is frequently precluded by difficulties in defining appropriate tumor boundaries. Adjuvant radiotherapy should be considered if an incomplete resection was carried out, especially in World Health Organization grade III neoplasms.


Assuntos
Neoplasias do Ventrículo Cerebral/cirurgia , Ganglioglioma/cirurgia , Hidrocefalia/fisiopatologia , Hipertensão Intracraniana/fisiopatologia , Procedimentos Neurocirúrgicos , Adulto , Idoso , Neoplasias do Ventrículo Cerebral/diagnóstico por imagem , Neoplasias do Ventrículo Cerebral/patologia , Neoplasias do Ventrículo Cerebral/fisiopatologia , Feminino , Estado Funcional , Ganglioglioma/diagnóstico por imagem , Ganglioglioma/patologia , Ganglioglioma/fisiopatologia , Humanos , Imageamento por Ressonância Magnética , Masculino , Mortalidade , Neoplasia Residual , Radioterapia Adjuvante , Tomografia Computadorizada por Raios X , Resultado do Tratamento
10.
Neuroimage ; 232: 117899, 2021 05 15.
Artigo em Inglês | MEDLINE | ID: mdl-33631332

RESUMO

Disproportionately enlarged subarachnoid-space hydrocephalus (DESH), characterized by tight high convexity CSF spaces, ventriculomegaly, and enlarged Sylvian fissures, is thought to be an indirect marker of a CSF dynamics disorder. The clinical significance of DESH with regard to cognitive decline in a community setting is not yet well defined. The goal of this work is to determine if DESH is associated with cognitive decline. Participants in the population-based Mayo Clinic Study of Aging (MCSA) who met the following criteria were included: age ≥ 65 years, 3T MRI, and diagnosis of cognitively unimpaired or mild cognitive impairment at enrollment as well as at least one follow-up visit with cognitive testing. A support vector machine based method to detect the DESH imaging features on T1-weighted MRI was used to calculate a "DESH score", with positive scores indicating a more DESH-like imaging pattern. For the participants who were cognitively unimpaired at enrollment, a Cox proportional hazards model was fit with time defined as years from enrollment to first diagnosis of mild cognitive impairment or dementia, or as years to last known cognitively unimpaired diagnosis for those who did not progress. Linear mixed effects models were fit among all participants to estimate annual change in cognitive z scores for each domain (memory, attention, language, and visuospatial) and a global z score. For all models, covariates included age, sex, education, APOE genotype, cortical thickness, white matter hyperintensity volume, and total intracranial volume. The hazard of progression to cognitive impairment was an estimated 12% greater for a DESH score of +1 versus -1 (HR 1.12, 95% CI 0.97-1.31, p = 0.11). Global and attention cognition declined 0.015 (95% CI 0.005-0.025) and 0.016 (95% CI 0.005-0.028) z/year more, respectively, for a DESH score of +1 vs -1 (p = 0.01 and p = 0.02), with similar, though not statistically significant DESH effects in the other cognitive domains. Imaging features of disordered CSF dynamics are an independent predictor of subsequent cognitive decline in the MCSA, among other well-known factors including age, cortical thickness, and APOE status. Therefore, since DESH contributes to cognitive decline and is present in the general population, identifying individuals with DESH features may be important clinically as well as for selection in clinical trials.


Assuntos
Encéfalo/diagnóstico por imagem , Ventrículos Cerebrais/diagnóstico por imagem , Disfunção Cognitiva/diagnóstico por imagem , Progressão da Doença , Hidrocefalia/diagnóstico por imagem , Imageamento por Ressonância Magnética/métodos , Espaço Subaracnóideo/diagnóstico por imagem , Idoso , Idoso de 80 Anos ou mais , Encéfalo/fisiologia , Ventrículos Cerebrais/fisiologia , Líquido Cefalorraquidiano/fisiologia , Pressão do Líquido Cefalorraquidiano/fisiologia , Disfunção Cognitiva/fisiopatologia , Estudos de Coortes , Feminino , Seguimentos , Humanos , Hidrocefalia/fisiopatologia , Estudos Longitudinais , Masculino , Valor Preditivo dos Testes , Fluxo Pulsátil/fisiologia , Espaço Subaracnóideo/fisiologia
11.
World Neurosurg ; 149: e931-e934, 2021 05.
Artigo em Inglês | MEDLINE | ID: mdl-33508492

RESUMO

OBJECTIVE: Post-traumatic hydrocephalus (PTH) is a common complication of craniocerebral injury. If not diagnosed in time, PTH can lead to clinical deterioration and a poor prognosis. The early diagnosis of PTH can lead to success with early treatment. However, PTH can be easily ignored during rehabilitation. The main purpose of the present study was to investigate whether plasma S100B protein levels can be used as a biochemical predictive index of PTH. We also explored the correlation among S100B protein levels, intracranial pressure, and PTH severity. METHODS: The data from 235 patients with traumatic brain injury treated from June 2014 to June 2019 in our hospital were retrospectively analyzed. Statistical analysis was performed on 3 serum S100B samples from each patient. The first sample was taken 1-3 days after the injury and surgery. The second sample was harvested during the stable period after treatment, and the third sample was taken when PTH had been confirmed by computed tomography. We analyzed the change in S100B protein levels, and intracranial pressure was measured by lumbar puncture. RESULTS: A total of 235 patients (Glasgow coma scale score <12) with traumatic brain injury were investigated. Of these 235 patients, 46 (19%) had developed PTH. The first and second S100B samples showed no significant differences between the patients with and without PTH. In the third sample, the S100B level of the patients with PTH was significantly greater than that of the patients without PTH, with a statistically significant difference. Statistical analysis found no correlation between the S100B level and the severity of PTH. CONCLUSIONS: Measurements of serum S100B can be used to predict for PTH. We found a positive correlation between S100B levels and intracranial pressure but no correlation with the severity of PTH. Thus, serum S100B could have important clinical significance for the early detection and evaluation of PTH.


Assuntos
Lesões Encefálicas Traumáticas/sangue , Hidrocefalia/sangue , Subunidade beta da Proteína Ligante de Cálcio S100/sangue , Adolescente , Adulto , Idoso , Idoso de 80 Anos ou mais , Lesões Encefálicas Traumáticas/complicações , Lesões Encefálicas Traumáticas/fisiopatologia , Diagnóstico Precoce , Feminino , Humanos , Hidrocefalia/diagnóstico , Hidrocefalia/etiologia , Hidrocefalia/fisiopatologia , Pressão Intracraniana , Masculino , Pessoa de Meia-Idade , Prognóstico , Índice de Gravidade de Doença , Adulto Jovem
12.
Nat Commun ; 12(1): 447, 2021 01 19.
Artigo em Inglês | MEDLINE | ID: mdl-33469018

RESUMO

Cerebrospinal fluid (CSF) provides vital support for the brain. Abnormal CSF accumulation, such as hydrocephalus, can negatively affect perinatal neurodevelopment. The mechanisms regulating CSF clearance during the postnatal critical period are unclear. Here, we show that CSF K+, accompanied by water, is cleared through the choroid plexus (ChP) during mouse early postnatal development. We report that, at this developmental stage, the ChP showed increased ATP production and increased expression of ATP-dependent K+ transporters, particularly the Na+, K+, Cl-, and water cotransporter NKCC1. Overexpression of NKCC1 in the ChP resulted in increased CSF K+ clearance, increased cerebral compliance, and reduced circulating CSF in the brain without changes in intracranial pressure in mice. Moreover, ChP-specific NKCC1 overexpression in an obstructive hydrocephalus mouse model resulted in reduced ventriculomegaly. Collectively, our results implicate NKCC1 in regulating CSF K+ clearance through the ChP in the critical period during postnatal neurodevelopment in mice.


Assuntos
Líquido Cefalorraquidiano/metabolismo , Plexo Corióideo/patologia , Hidrocefalia/patologia , Membro 2 da Família 12 de Carreador de Soluto/metabolismo , Animais , Animais Recém-Nascidos , Plexo Corióideo/diagnóstico por imagem , Plexo Corióideo/crescimento & desenvolvimento , Plexo Corióideo/metabolismo , Dependovirus/genética , Modelos Animais de Doenças , Embrião de Mamíferos , Feminino , Vetores Genéticos/administração & dosagem , Vetores Genéticos/genética , Humanos , Hidrocefalia/congênito , Hidrocefalia/diagnóstico , Hidrocefalia/fisiopatologia , Injeções Intraventriculares , Pressão Intracraniana/fisiologia , Imageamento por Ressonância Magnética , Masculino , Camundongos , Camundongos Transgênicos , Membro 2 da Família 12 de Carreador de Soluto/genética
13.
World Neurosurg ; 148: 24-28, 2021 04.
Artigo em Inglês | MEDLINE | ID: mdl-33444826

RESUMO

BACKGROUND: Schwannomas at the craniocervical junction commonly originate from the lower cranial nerves or C1 and C2 nerves. To date, very few cases of C1 schwannomas have been described in the literature, and the majority involve either the intra- or the extradural compartment, but not both. To our knowledge, this report documents the first case of a dumbbell-shaped C1 schwannoma that encompassed both intra- and extradural compartments and was accompanied by hydrocephalus. CASE DESCRIPTION: The patient was admitted to our hospital, where magnetic resonance imaging revealed a tumor at the craniocervical junction, extending from the C1 level of the right first cervical vertebra into the cerebellopontine angle with 2 giant cysts. We removed the tumor by performing a midline posterior craniectomy and cervical laminectomy. Intraoperatively, the tumor was found to originate from the right C1 posterior root. The pathological diagnosis was of a schwannoma. The patient was subsequently discharged without any neurologic deficits. CONCLUSIONS: To our knowledge, we present the first case of a dumbbell-shaped C1 schwannoma with intracranial extensions and accompanying hydrocephalus. The tumor had spread inside and outside the dura, but was safely removed. Our findings in this case emphasize that to achieve safe resection, detailed case-specific preoperative consideration is essential.


Assuntos
Hidrocefalia/diagnóstico por imagem , Neurilemoma/diagnóstico por imagem , Neoplasias do Sistema Nervoso Periférico/diagnóstico por imagem , Raízes Nervosas Espinhais , Anorexia/fisiopatologia , Ângulo Cerebelopontino , Angiografia Cerebral , Atlas Cervical , Vértebras Cervicais/cirurgia , Angiografia por Tomografia Computadorizada , Craniotomia , Feminino , Transtornos Neurológicos da Marcha/fisiopatologia , Humanos , Hidrocefalia/etiologia , Hidrocefalia/fisiopatologia , Hidrocefalia/cirurgia , Laminectomia , Pessoa de Meia-Idade , Náusea/fisiopatologia , Neurilemoma/complicações , Neurilemoma/fisiopatologia , Neurilemoma/cirurgia , Neoplasias do Sistema Nervoso Periférico/complicações , Neoplasias do Sistema Nervoso Periférico/fisiopatologia , Neoplasias do Sistema Nervoso Periférico/cirurgia
14.
Brain Res ; 1753: 147229, 2021 02 15.
Artigo em Inglês | MEDLINE | ID: mdl-33385374

RESUMO

Hydrocephalus associated with long term spaceflight (HALS) for missions lasting over five months is well described but poorly understood. While structural changes of the brain due to microgravitational forces affecting the circulation of cerebrospinal fluid (CSF) have been described as one potential cause, we propose an alternative hypothesis based on dynamic disequilibrium of macromolecular transport across the blood brain barrier. We propose that factors altering physiology under conditions of spaceflight such as microgravity, hypercapnia, venous hypertension, medications, and dietary substances contribute to increased protein load in the ventricles and/or contribute to impairment of transport out of the ventricles that results in HALS. Individual variation in the genetic expression of efflux transporters (p-glycoprotein) has been shown to correlate with the presence and degree of hydrocephalus in animal studies. We describe the evidence behind this concept and propose how these factors can be studied in order to determine the underlying pathogenesis which is imperative in order to cure or prevent HALS.


Assuntos
Encéfalo/fisiopatologia , Hidrocefalia/patologia , Hidrocefalia/fisiopatologia , Voo Espacial , Animais , Encéfalo/patologia , Ventrículos Cerebrais/patologia , Ventrículos Cerebrais/fisiopatologia , Humanos , Transtornos Psicomotores/complicações , Transtornos Psicomotores/fisiopatologia , Tempo
15.
Anat Sci Int ; 96(1): 87-96, 2021 Jan.
Artigo em Inglês | MEDLINE | ID: mdl-32789737

RESUMO

Cerebellar abnormalities are commonly associated with hydrocephalus. However, the effect of hydrocephalus on the otherwise normal cerebellum has been largely neglected. This study assesses the morphological changes in the Purkinje cells in relation to cerebellar dysfunction observed in juvenile hydrocephalic rats. Fifty-five three-week old albino Wistar rats were used, hydrocephalus was induced by intracisternal injection of kaolin (n = 35) and others served as controls (n = 20). Body weight measurements, hanging wire, negative geotaxis, and open field tests were carried out at the onset and then weekly for 4 weeks, rats were killed, and their cerebella processed for Hematoxylin and Eosin, Cresyl violet and Golgi staining. Qualitative and quantitative studies were carried out; quantitative data were analyzed using two-way ANOVA and independent T tests at p < 0.05. Hydrocephalic rats weighed less than controls (p = 0.0247) but their cerebellar weights were comparable. The hydrocephalic rats had a consistently shorter latency to fall in the hanging wire test (F(4,112) = 18.63; p < 0.0001), longer latency to turn in the negative geotaxis test (F(4,112) = 22.2; p < 0.0001), and decreased horizontal (F(4,112) = 4.172, p = 0.0035) and vertical movements (F(4,112) = 4.397; p = 0.0024) in the open field test than controls throughout the 4 weeks post-induction. Cellular compression in the granular layer, swelling of Purkinje cells with vacuolations, reduced dendritic arborization and increased number of pyknotic Purkinje cells were observed in hydrocephalic rats. Hydrocephalus caused functional and morphological changes in the cerebellar cortex. Purkinje cell loss, a major pathological feature of hydrocephalus, may be responsible for some of the motor deficits observed in this condition.


Assuntos
Cerebelo/patologia , Cerebelo/fisiopatologia , Hidrocefalia/patologia , Hidrocefalia/fisiopatologia , Caulim/efeitos adversos , Desempenho Psicomotor , Células de Purkinje/fisiologia , Animais , Cerebelo/citologia , Modelos Animais de Doenças , Hidrocefalia/induzido quimicamente , Movimento , Ratos Wistar
16.
World Neurosurg ; 146: e1054-e1062, 2021 02.
Artigo em Inglês | MEDLINE | ID: mdl-33246177

RESUMO

OBJECTIVE: Coil embolization with Y stenting is recognized as a suitable treatment for complex wide-necked aneurysms. Y stenting comprises crossing-Y stenting, in which a stent is passed through the interstices of another stent, and kissing-Y stenting, in which 2 stents are arranged in parallel. The purpose of this study was to elucidate how to distinguish between use of the 2 Y-stenting techniques. METHODS: Clinical and angiographic data of patients who underwent coil embolization with Y stenting at our department from 2015 to 2019 were retrospectively analyzed. Basic characteristics, endovascular procedure, complications, and outcomes were compared between kissing-Y and crossing-Y stenting groups. RESULTS: Thirty-eight intracranial aneurysms in 38 consecutive patients were included in this study. Nineteen patients (50%) were treated with coil embolization with kissing-Y stenting and 19 (50%) with crossing-Y. Endovascular procedures were successfully performed in all but 1 patient, in the kissing-Y group, who had stent migration. One hemorrhage (2.6%) recurred 12 months after coiling with kissing-Y stenting. Angiographic follow-up (mean, 15.8 months) was available in 35 patients. Adequate occlusion was shown in 14 patients (77.8%) and 13 patients (76.5%) in the kissing-Y and crossing-Y groups, respectively. Larger, wider-necked, and more proximal aneurysms were treated with kissing-Y stenting than with crossing-Y stenting, although there were no significant differences between the groups in complication rates or clinical outcomes. CONCLUSIONS: Kissing-Y and crossing-Y stenting of intracranial aneurysms were both feasible and yielded reasonable angiographic and clinical results. The choice between the kissing-Y or crossing-Y-stenting technique should be decided according to the angioarchitecture of targeted aneurysms.


Assuntos
Aneurisma Roto/cirurgia , Procedimentos Endovasculares/métodos , Aneurisma Intracraniano/cirurgia , Stents , Hemorragia Subaracnóidea/cirurgia , Idoso , Idoso de 80 Anos ou mais , Doenças Assintomáticas , Angiografia Cerebral , Embolização Terapêutica , Feminino , Cefaleia/fisiopatologia , Humanos , Hidrocefalia/fisiopatologia , Aneurisma Intracraniano/fisiopatologia , Masculino , Pessoa de Meia-Idade , Transtornos da Motilidade Ocular/fisiopatologia , Falha de Prótese , Estudos Retrospectivos , Transtornos da Visão/fisiopatologia
17.
World Neurosurg ; 146: e1071-e1078, 2021 02.
Artigo em Inglês | MEDLINE | ID: mdl-33246179

RESUMO

OBJECTIVE: Colloid cysts (CCs) are histologically benign lesions that carry the risk of causing obstructive hydrocephalus. The indication for surgery for symptomatic cysts is unquestioned; however, the management of incidentally detected lesions has remained controversial. We independently evaluated the CC risk score (CCRS) as a useful risk stratification scheme. METHODS: The demographics, imaging characteristics, and clinical presentations of 103 patients with CC during a 10-year period were correlated with symptoms and the occurrence of hydrocephalus. The discriminate capacity of the CCRS was quantified and a decision algorithm formulated. RESULTS: The correlates of symptoms included age <65 years, diameter ≥7 mm, anterior location, hyperintensity on T2-weighted fluid-attenuated inversion recovery (FLAIR) and/or T2-weighted images, and headache at presentation without an alternative explanation. On multivariate analysis, age <65 years, diameter ≥7 mm, and headache at presentation predicted for symptomatic cysts. The CCRS was highly effective at identifying symptomatic patients and those with obstructive hydrocephalus. All patients with cyst growth and symptomatic progression had had an initial CCRS of ≥3 plus hyperintensity (preexisting or developing) on T2-weighted FLAIR images. CONCLUSIONS: The CCRS performed exceptionally well in distinguishing the highest risk patients. Closer follow-up is recommended for younger asymptomatic patients with cysts near the foramen of Monro. Hyperintensity on T2-weighted FLAIR images might indicate greater growth potential for small lesions.


Assuntos
Cistos Coloides/cirurgia , Hidrocefalia/cirurgia , Adulto , Fatores Etários , Idoso , Doenças Assintomáticas , Tomada de Decisão Clínica , Cistos Coloides/complicações , Cistos Coloides/diagnóstico por imagem , Cistos Coloides/fisiopatologia , Gerenciamento Clínico , Progressão da Doença , Feminino , Cefaleia/fisiopatologia , Humanos , Hidrocefalia/etiologia , Hidrocefalia/fisiopatologia , Achados Incidentais , Imageamento por Ressonância Magnética , Masculino , Pessoa de Meia-Idade , Análise Multivariada , Procedimentos Neurocirúrgicos , Medição de Risco , Tomografia Computadorizada por Raios X , Adulto Jovem
18.
Ann Clin Transl Neurol ; 8(2): 523-524, 2021 02.
Artigo em Inglês | MEDLINE | ID: mdl-33338330

RESUMO

A 61-year-old man with past medical history significant for prediabetes, hyperlipidemia and high-grade prostate intraepithelial neoplasia presents with headaches for one month. Imaging of his brain reveals hydrocephalus and spine imaging reveals a cord lesion. These findings are discussed further in the case.


Assuntos
Cefaleia , Hidrocefalia/complicações , Hidrocefalia/fisiopatologia , Hemorragia Subaracnóidea/complicações , Hemorragia Subaracnóidea/fisiopatologia , Cefaleia/diagnóstico , Cefaleia/etiologia , Cefaleia/fisiopatologia , Humanos , Hidrocefalia/diagnóstico por imagem , Imageamento por Ressonância Magnética , Masculino , Pessoa de Meia-Idade , Neoplasia Prostática Intraepitelial , Neoplasias da Próstata , Neoplasias da Medula Espinal/diagnóstico , Neoplasias da Medula Espinal/fisiopatologia , Hemorragia Subaracnóidea/diagnóstico por imagem
19.
Ann Clin Lab Sci ; 50(6): 818-824, 2020 Nov.
Artigo em Inglês | MEDLINE | ID: mdl-33334799

RESUMO

Baraitser-Winter Cerebro-fronto-facial syndrome (BWCFF, OMIM #243310, #614583) is caused by a heterozygous gain-of-function mutation of ACTB and ACTG1 that encodes actin. The syndrome is characterized by striking facial features, structural brain abnormalities, ocular coloboma, hearing loss, cardiac defects, intellectual disabilities, short stature, and developmental delay. We report a two-year-old girl who had distinctive facial features, including hypertelorism, arched eyebrows, bilateral ptosis, short broad nose with a flat nasal tip, long philtrum, retrognathia, low-set ears, and a thin upper lip. In addition, she also exhibited short stature, pectus excavatum, developmental delay, brain malformation, and hearing loss. Targeted gene panel sequencing identified a de novo heterozygous missense variant c.826G>A (p.Glu276Lys) in ACTB This is the first Korean case of BWCFF with a novel mutation in ACTB.


Assuntos
Actinas/genética , Transtornos do Crescimento/genética , Hidrocefalia/genética , Retardo Mental Ligado ao Cromossomo X/genética , Obesidade/genética , Anormalidades Múltiplas/genética , Actinas/metabolismo , Pré-Escolar , Coloboma/genética , Deficiências do Desenvolvimento/genética , Face , Facies , Feminino , Transtornos do Crescimento/fisiopatologia , Heterozigoto , Humanos , Hidrocefalia/fisiopatologia , Deficiência Intelectual/genética , Retardo Mental Ligado ao Cromossomo X/fisiopatologia , Obesidade/fisiopatologia , República da Coreia , Síndrome
20.
PLoS One ; 15(11): e0241974, 2020.
Artigo em Inglês | MEDLINE | ID: mdl-33186351

RESUMO

Neuroradiological abnormalities in tuberculous meningitis (TBM) are common, but the exact relationship with clinical and inflammatory markers has not been well established. We performed magnetic resonance imaging (MRI) at baseline and after two months treatment to characterise neuroradiological patterns in a prospective cohort of adult TBM patients in Indonesia. We included 48 TBM patients (median age 30, 52% female, 8% HIV-infected), most of whom had grade II (90%), bacteriologically confirmed (71%) disease, without antituberculotic resistance. Most patients had more than one brain lesion (83%); baseline MRIs showed meningeal enhancement (89%), tuberculomas (77%), brain infarction (60%) and hydrocephalus (56%). We also performed an exploratory analysis associating MRI findings to clinical parameters, response to treatment, paradoxical reactions and survival. The presence of multiple brain lesion was associated with a lower Glasgow Coma Scale and more pronounced motor, lung, and CSF abnormalities (p-value <0.05). After two months, 33/37 patients (89%) showed worsening of MRI findings, mostly consisting of new or enlarged tuberculomas. Baseline and follow-up MRI findings and paradoxical responses showed no association with six-month mortality. Severe TBM is characterized by extensive MRI abnormalities at baseline, and frequent radiological worsening during treatment.


Assuntos
Encéfalo/fisiologia , Encéfalo/fisiopatologia , Tuberculose Meníngea/fisiopatologia , Adolescente , Adulto , Antituberculosos/uso terapêutico , Infarto Encefálico/fisiopatologia , Método Duplo-Cego , Feminino , Escala de Coma de Glasgow , Humanos , Hidrocefalia/fisiopatologia , Imageamento por Ressonância Magnética/métodos , Masculino , Pessoa de Meia-Idade , Estudos Prospectivos , Tuberculose Meníngea/tratamento farmacológico , Adulto Jovem
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