Microcephaly (MC), previously considered rare, is now a health emergency of international concern because of the devastating Zika virus pandemic outbreak of 2015. The authors describe the electroencephalogram (EEG) findings in sleep EEG of epileptic children who were born with microcephaly in areas of Brazil with active Zika virus transmission between 2014 and 2017. The authors reviewed EEGs from 23 children. Nine were females (39.2%), and the age distribution varied from 4 to 48 months. MC was associated with mother positive serology to toxoplasmosis (toxo), rubella (rub), herpes, and dengue (1 case); toxo (1 case); chikungunya virus (CHIKV) (1 case); syphilis (1 case); and Zika virus (ZIKV) (10 cases). In addition, 1 case was associated with perinatal hypoxia and causes of 9 cases remain unknown. The main background EEG abnormality was diffuse slowing (10 cases), followed by classic (3 cases) and modified (5 cases) hypsarrhythmia. A distinct EEG pattern was seen in ZIKV (5 cases), toxo (2 cases), and undetermined cause (1 case). It was characterized by runs of frontocentrotemporal 4.5-13 Hz activity (7 cases) or diffuse and bilateral runs of 18-24 Hz (1 case). In ZIKV, this rhythmic activity was associated with hypsarrhythmia or slow background. Further studies are necessary to determine if this association is suggestive of ZIKV infection. The authors believe that EEG should be included in the investigation of all newly diagnosed congenital MC, especially those occurring in areas of autochthonous transmission of ZIKV.
Objective Rehabilitation top‐down techniques based on brain stimulation present variable outcomes in unilateral spatial neglect (USN) after stroke. This study aimed to examine the effects of physical therapy after anodal and cathodal transcranial direct current stimulation (A‐tDCS and C‐tDCS, respectively) to improve visuospatial and functional impairments in individuals with USN after stroke. Methods This double‐blinded, pilot randomized clinical trial enrolled patients with USN after ischemic stroke. Randomization was stratified according to the Behavior Inattention Test–Conventional (BIT‐C) and Catherine Bergego Scale (CBS). Outpatient physical therapy was conducted for 7.5 weeks after 20 minutes of tDCS. The primary outcome was the USN degree evaluated by the BIT‐C. Secondary outcomes were the difference in CBS score, stroke severity (National Institutes of Health Stroke Scale [NIHSS]), disability (modified Rankin Scale), autonomy (Barthel Index, Functional Independence Measure), and quality of life (EuroQol Group 5‐Dimension Self‐Report Questionnaire). Outcomes were analyzed using an analysis of covariance model corrected by age, baseline NIHSS, and baseline BIT‐C. Pairwise post hoc comparisons were performed using Bonferroni correction. Results In the primary outcomes, A‐tDCS led to greater improvement in BIT‐C after intervention (mean difference [MD] = 18.4, 95% confidence interval [CI] = 3.9–32.8, p = 0.008) compared to sham. However, no significant differences were observed between A‐tDCS and C‐tDCS (MD = 13.9, 95% CI = −0.3 to 28.1, p = 0.057), or C‐tDCS and sham (MD = 4.5, 95% CI = −9.7 to 18.8, p = 0.99). There were no significant differences between groups in terms of secondary outcomes. Interpretation A‐tDCS associated with physical therapy can decrease the severity of USN after stroke. However, these preliminary findings must be confirmed by collecting additional evidence in a larger phase 3 trial. ANN NEUROL 2022;92:400–410
There is a high demand for stroke rehabilitation in the Brazilian public health system, but most studies that have addressed rehabilitation for unilateral spatial neglect (USN) after stroke have been performed in high-income countries. Therefore, the aim of this study was to analyze USN patient recruitment in a multicenter noninvasive brain stimulation clinical trial performed in Brazil and to provide study design recommendations for future studies. We evaluated the reasons for exclusion of patients from a multicenter, randomized, double-blinded clinical trial of rehabilitation of USN patients after stroke. Clinical and demographic variables were compared between the included and excluded patients. A descriptive statistical analysis was performed. Only 173 of the 1953 potential neglect patients (8.8%) passed the initial screening. After screening evaluation, 87/173 patients (50.3%) were excluded for clinical reasons. Cognitive impairment led to the exclusion of 21/87 patients (24.1%). Low socioeconomic status led to the exclusion of 37/173 patients (21.4%). Difficulty obtaining transportation to access treatment was the most common reason for their exclusion (16/37 patients, 43.3%). The analyzed Brazilian institutions have potential for conducting studies of USN. The recruitment of stroke survivors with USN was restricted by the study design and limited financial support. A history of cognitive impairment, intracranial stenting or craniectomy, and lack of transportation were the most common barriers to participating in a multicenter noninvasive brain stimulation trial among patients with USN after stroke.
Case presentation: We describe the case of a 75-year-old female, with no past relevant comorbidities. The onset was of mental confusion and imbalance, within two weeks progressing to dysphonia, dysphagia and spastic quadriparesis. Brain MRI showed hyperintense T2/FLAIR (T2-weighted-FluidAttenuated Inversion Recovery) lesions in white matter, with cortical sparing, restricted diffusion and gadolinium enhancement. Demyelinating diseases and central nervous system lymphoma hypothesis were made. Two months later a brain biopsy was performed. Diffuse white matter vacuolar impairment was found and no inflammatory infiltrate, resembling Canavan’s disease. The patient had progressive clinical worsening, with prolonged hospitalization and poor general condition at discharge, not enduring further investigation. She deceased after 8 months of symptoms onset. Discussion: The diagnosis of acute leukoencephalopathies is challenging and neuroimaging may be helpful. Diffusion, contrast enhancement and corticosubcortical relation in magnetic resonance imaging (MRI) can present valuable clues. In this case, for example, acute disseminated encephalomyelitis was less likely due to the MRI evidencing dissemination in time. To better address malignancy, brain biopsy was mandatory. Not only cancer was excluded, but the demyelination was revealed to possibly be neurodegenerative, which was neither clinically nor radiographically evident. The vacuolar pattern found is described in Canavan disease, a rare leukoencephalopathy with onset at 1–4 months age, and mean survival of months to few years. In literature there is only another report, cataloged in our Pathology department, of a 43-yearold male with similar clinical, imaging and histological findings. This case may illustrate a not yet cataloged disease, maybe an unknown presentation of Canavan disease spectrum, adding one more differential diagnosis for acute leukoencephalopathies.
We report a case of a child whose EEG demonstrated extreme spindles (ES) after acute lymphoblastic leukemia treatment. This finding has not been reported previously. In 1962, Gibbs and Gibbs described the ES EEG pattern due to its high amplitude (200 to 400 μV). ES are a rare spindle variant that is found in EEGs of 0.05% of normal children (average age, 3 years, with a range of 1 to 12 years), and are even rarer after 11 years. Moreover due to changes in the white matter of the frontal lobe, ES have been associated with such conditions as cerebral palsy and mental retardation, residual brain damage, undefined infections, infantile neuroaxonal dystrophy, Menkes' kinky-hair syndrome, congenital muscular dystrophy, hydrocephalus, porencephaly, epilepsy, progressive cerebellar degeneration, and mycoplasma encephalitis. Methotrexate has a notably toxic effect on the central nervous system, with leukoencephalopathy being the most common form. In our case, frontocentral ES were associated with hyperintense lesions in the white matter of the frontal lobe. Lesional deafferentation can be the substrate for an almost continuous ES, since both initiation and termination of spindle oscillations are thought to originate in thalamocortical neurons. Thus, we postulate that in some cases a partial functional cortical differentiation could generate ES.
Background This study evaluated histopathological characteristics of bone marrow (BM) of patients with immune thrombocytopenic purpura (ITP) and sought to find possible associations among them and clinical aspects. Method A retrospective study was carried out with 120 patients using BM clot and BM biopsy samples, including morphological (cytological and histological) re-evaluation, reticulin and hemosiderin analysis, and clinical outcome review of medical records. Immunohistochemistry (CD34 and CD117) was applied in a group of patients with increased reticulin, with the objective of exclusion Myelodysplastic syndrome cases Results Megakaryocytic hyperplasia was observed in 109 (90.8%) patients and increased reticulin was diagnosed in nine patients, five of them with a clinically unfavorable outcome (p = 0.042). The increase in reticulin graduation was associated with a higher risk of an unfavorable outcome. Conclusion Increased reticulin degree in BM of patients with ITP is associated with an unfavorable outcome in this study. It is rarely explored in the literature and may provide information that contributes to understanding the patient's outcomes.
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