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- Multidisciplinary Approaches and Molecular Diagnostics in New-Onset Refractory Status Epilepticus and Nontuberculous Mycobacterial CNS Infections in the ICU: A Case Report.Publication . Batarda Sena, Pedro Manuel; Ferro, Margarida; Alves Cabrita, Joana; Pontinha, Carlos; Mestre, Ana; Oliveira, Mário; Bento, LuisThe diagnosis and management of complex neurological conditions such as New-Onset Refractory Status Epilepticus (NORSE) and central nervous system (CNS) infections caused by non-tuberculous mycobacteria (NTM) pose substantial difficulties in intensive care units (ICUs). This article combines a case report and a literature review that explores the diagnostic dilemmas and therapeutic strategies for these critical conditions. We report the case of an 83-year-old female with chronic granulomatous meningitis secondary to NTM, presenting a challenging diagnostic and complex management complexity typical of such a rare disease through a period time of five years. Her case emphasized the importance of a multidisciplinary approach in such cases, as the interplay between neurology and intensive care was vital. The need for new molecular diagnostic technologies is shown to be of high significance in identifying the causative pathogens and improving patient outcomes in these rare but critical conditions.
- Prognostic Factors Associated with Disability in a Cohort of Neuromyelitis Optica Spectrum Disorder and MOG-Associated Disease from a Nationwide Portuguese Registry.Publication . Moura, João; Samões, Raquel; Sousa, Ana Paula; Figueiroa, Sónia; Mendonça, Teresa; Abreu, Pedro; Guimarães, Joana; Melo, Claúdia; Sousa, Raquel; Soares, Mafalda; Correia, Ana Sofia; Marques, Inês Brás; Perdigão, Sandra; Alves, Ivânia; Felgueiras, Helena; Nzwalo, Hipólito; Mendes, Irene; Almeida, Vânia; Boleixa, Daniela; Carneiro, Paula; Neves, Esmeralda; Silva, Ana Martins; Sá, Maria José; Santos, ErnestinaIntroduction: Neuromyelitis optica spectrum disorders (NMOSD) and MOG-associated disease (MOGAD) are an increasingly recognized group of demyelinating disorders of the central nervous system. Previous studies suggest that prognosis is predicted by older age at onset, number of relapses, the severity of the first attack and autoantibody status. Objective: To study prognostic factors associated with disability progression and additional relapses in the 3-year follow-up of a national NMOSD/MOGAD cohort. Results: Out of 180 of the initial Portuguese cohort, data on 82 patients was available at the end of the follow-up period (2019-2022). Two patients died. Twenty (24.4%) patients had one or more attack in this period (25 attacks in total), mostly transverse myelitis (TM) (56.0%) or optic neuritis (32.0%). MOGAD was significantly associated with a monophasic disease course (p = 0.03), with milder attacks (p = 0.01), while AQP4 + NMOSD was associated with relapses (p = 0.03). The most common treatment modalities were azathioprine (38.8%) and rituximab (18.8%). AQP4 + NMOSD more frequently required chronic immunosuppressive treatment, particularly rituximab (p = 0.01). Eighteen (22.5%) had an EDSS ≥6 at the end of the follow-up. AQP4 + NMOSD (p < 0.01) and the occurrence of transverse myelitis (TM) during disease (p = 0.04) correlated with an EDSS≥6 at the end of the follow-up period. MOGAD was significantly associated with an EDSS<6 (p < 0.01), and MOG+ cases that reached an EDSS>6 were significantly older (64.0 ± 2.8 versus 31.0 ± 17.1, p = 0.017). A bivariate logistic regression model including the serostatus and TM attacks during disease history successfully predicted 72.2% of patients that progressed to an EDSS≥6. Conclusion: This study highlights that myelitis predict increased disability (EDSS≥6) in NMOSD/MOGAG and AQP4 positivity is associated with increased disability.
- Cerebral Venous Sinus Thrombosis in Traumatic Brain Injury: a Systematic Review of its Complications, Effect on Mortality, Diagnostic and Therapeutic Management, and Follow-Up.Publication . Netteland, Dag Ferner; Sandset, Else Charlotte; Mejlænder-Evjensvold, Magnus; Aarhus, Mads; Jeppesen, Elisabeth; Aguiar de Sousa, Diana; Helseth, Eirik; Brommeland, Tor; FrontiersObjective: Cerebral venous sinus thrombosis (CVST) is increasingly being recognized in the setting of traumatic brain injury (TBI), but its effect on TBI patients and its management remains uncertain. Here, we systematically review the currently available evidence on the complications, effect on mortality and the diagnostic and therapeutic management and follow-up of CVST in the setting of TBI. Methods: Key clinical questions were posed and used to define the scope of the review within the following topics of complications; effect on mortality; diagnostics; therapeutics; recanalization and follow-up of CVST in TBI. We searched relevant databases using a structured search strategy. We screened identified records according to eligibility criteria and for information regarding the posed key clinical questions within the defined topics of the review. Results: From 679 identified records, 21 studies met the eligibility criteria and were included, all of which were observational in nature. Data was deemed insufficiently homogenous to perform meta-analysis and was narratively synthesized. Reported rates of venous infarctions ranged between 7 and 38%. One large registry study reported increased in-hospital mortality in CVSP and TBI compared to a control group with TBI alone in adjusted analyses. Another two studies found midline CVST to be associated with increased risk of mortality in adjusted analyses. Direct data to inform the optimum diagnostic and therapeutic management of the condition was limited, but some data on the safety, and effect of anticoagulation treatment of CVST in TBI was identified. Systematic data on recanalization rates to guide follow-up was also limited, and reported complete recanalization rates ranged between 41 and 86%. In the context of the identified data, we discuss the diagnostic and therapeutic management and follow-up of the condition. Conclusion: Currently, the available evidence is insufficient for evidence-based treatment of CVST in the setting of TBI. However, there are clear indications in the presently available literature that CVST in TBI is associated with complications and increased mortality, and this indicates that management options for the condition must be considered. Further studies are needed to confirm the effects of CVST on TBI patients and to provide evidence to support management decisions.
- Basilar Artery Occlusion Management: Specialist Perspectives From an International SurveyPublication . Edwards, C; Drumm, B; Siegler, J; Schonewille, W; Klein, P; Huo, X; Chen, Y; Abdalkader, M; Qureshi, M; Strbian, D; Liu, X; Hu, W; Ji, X; Li, C; Fischer, U; Nagel, S; Puetz, V; Michel, P; Alemseged, F; Sacco, S; Yamagami, H; Yaghi, S; Strambo, D; Kristoffersen, E; Sandset, E; Mikulik, R; Tsivgoulis, G; Masoud, H; Aguiar de Sousa, D; Marto, JP; Lobotesis, K; Roi, D; Berberich, A; Demeestere, J; Meinel, T; Rivera, R; Poli, S; Ton, M; Zhu, Y; Li, F; Sang, H; Thomalla, G; Parsons, M; Campbell, B; Zaidat, O; Chen, HS; Field, T; Raymond, J; Kaesmacher, J; Nogueira, R; Jovin, T; Sun, D; Liu, R; Qureshi, A; Qiu, Z; Miao, Z; Banerjee, S; Nguyen, TBackground and purpose: Two early basilar artery occlusion (BAO) randomized controlled trials did not establish the superiority of endovascular thrombectomy (EVT) over medical management. While many providers continue to recommend EVT for acute BAO, perceptions of equipoise in randomizing patients with BAO to EVT versus medical management may differ between clinician specialties. Methods: We conducted an international survey (January 18, 2022 to March 31, 2022) regarding management strategies in acute BAO prior to the announcement of two trials indicating the superiority of EVT, and compared responses between interventionalists (INTs) and non-interventionalists (nINTs). Selection practices for routine EVT and perceptions of equipoise regarding randomizing to medical management based on neuroimaging and clinical features were compared between the two groups using descriptive statistics. Results: Among the 1245 respondents (nINTs = 702), INTs more commonly believed that EVT was superior to medical management in acute BAO (98.5% vs. 95.1%, p < .01). A similar proportion of INTs and nINTs responded that they would not randomize a patient with BAO to EVT (29.4% vs. 26.7%), or that they would only under specific clinical circumstances (p = .45). Among respondents who would recommend EVT for BAO, there was no difference in the maximum prestroke disability, minimum stroke severity, or infarct burden on computed tomography between the two groups (p > .05), although nINTs more commonly preferred perfusion imaging (24.2% vs. 19.7%, p = .04). Among respondents who indicated they would randomize to medical management, INTs were more likely to randomize when the National Institutes of Health Stroke Scale was ≥10 (15.9% vs. 6.9%, p < .01). Conclusions: Following the publication of two neutral clinical trials in BAO EVT, most stroke providers believed EVT to be superior to medical management in carefully selected patients, with most indicating they would not randomize a BAO patient to medical treatment. There were small differences in preference for advanced neuroimaging for patient selection, although these preferences were unsupported by clinical trial data at the time of the survey.
- A Case of Trigeminal Malignant Melanotic Nerve Sheath Tumor in the Wide Spectrum of Melanotic and Nerve Sheath TumorsPublication . Rachão, A; Ferro, M; Roque, R; Rainha Campos, A; Pimentel, J
- Postictal Psychosis: Case Report and Literature ReviewPublication . Regala, J; Lourenço, J; Moniz-Pereira, F; Bento, APostictal psychosis (PIP) is one of the most common types of psychosis in epileptic patients. By virtue of the paucity of research on PIP, its pathophysiology remains not completely understood. Our case report describes a clinical picture of PIP, characterized by pleomorphic features, with neither Schneider's first-rank symptoms nor negative symptoms of schizophrenia, in a longstanding epileptic female patient with a history of nonadherence to antiepileptic treatment and poorly controlled seizures. Additionally, she had previous cognitive impairment and encephalomalacia in the right parietooccipital region as a sequela of a moderate-to-severe traumatic brain injury known to precede the emergence of the epilepsy. In light of our findings, we critically reviewed the current literature on postictal psychoses providing insight into its neurobiological underpinnings.
- Influence of Physicians’ Risk Perception on Switching Treatments Between High- Efficacy and Non–High-Efficacy Disease‑Modifying Therapies in Multiple SclerosisPublication . Seifer, G; Arun, T; Capela, C; Laureys, G; Jones, E; Dominguez-Castro, P; Sanchez-de la Rosa, R; Hiltl, S; Iaffaldano, PBackground: The decision of initiating treatment for multiple sclerosis (MS) with a high-efficacy DMT (HE DMT) or non-high-efficacy DMT (non-HE DMT) is influenced by several factors, including risk perception of patients and physicians. Objective: Investigate the influence of physicians' risk perception on decision-making when switching treatments for MS and the reasons for switching. Methods: Data were drawn from the Adelphi Real-World MS Disease-Specific Program (a retrospective survey) and analysis included people with RMS identified between 2017- 2021. Results: Of 4129 patients with reasons for switch available, 3538 switched from non-HE DMT and 591 from HE DMT. Overall, 4.7% of patients were switched treatment by their physicians due to the risk of malignancies and infections including PML risk. The proportion of switches that were made due to the risk of PML were 23.9% in the HE DMT and 0.5% in the non-HE DMT groups. The top reasons for switching were relapse frequency (non-HE DMT vs HE-DMT: 26.8% vs 15.2%), lack of efficacy (20.9 vs 11.7) and increased number of MRI lesions (20.3% vs 12.4%). Conclusions: Physicians' risk perception of malignancies and infection excluding PML was not a leading factor when switching treatment. The risk of PML was a key factor, especially for switching patients from HE DMTs. In both groups, lack of efficacy was the key contributing factor for switching. Initiating the treatment with HE DMTs may potentially reduce the number of switches due to sub-optimal efficacy. These findings might help physicians to engage more in discussions with patients about the benefit/risk profile of DMTs.
- Simplifying Data Analysis in Biomedical Research: an Automated, User-Friendly ToolPublication . Araújo, R; Ramalhete, L; Viegas, A; Von Rekowski, C; Fonseca, T; Calado, C; Bento, LRobust data normalization and analysis are pivotal in biomedical research to ensure that observed differences in populations are directly attributable to the target variable, rather than disparities between control and study groups. ArsHive addresses this challenge using advanced algorithms to normalize populations (e.g., control and study groups) and perform statistical evaluations between demographic, clinical, and other variables within biomedical datasets, resulting in more balanced and unbiased analyses. The tool's functionality extends to comprehensive data reporting, which elucidates the effects of data processing, while maintaining dataset integrity. Additionally, ArsHive is complemented by A.D.A. (Autonomous Digital Assistant), which employs OpenAI's GPT-4 model to assist researchers with inquiries, enhancing the decision-making process. In this proof-of-concept study, we tested ArsHive on three different datasets derived from proprietary data, demonstrating its effectiveness in managing complex clinical and therapeutic information and highlighting its versatility for diverse research fields.
- Contralateral Upper Limb Weakness Following Botulinum Toxin A Injection for Poststroke SpasticityPublication . Camões- Barbosa, A; Mendes Ribeiro, I; Medeiros, LBotulinum toxin type A has been approved for spasticity management in poststroke patients. The adverse effects are generally of two types: those related to local injection; and those related to the systemic effects from spread of the toxin. Contralateral weakness after botulinum toxin A treatment is a rarely reported adverse effect. We report the case of a 33-year-old female who had been receiving regular injections of incobotulinum toxin A due to spasticity of the right limbs after a hemorrhagic stroke. A switch was made to abobotulinum toxin A with an overall conversion ratio of 1:3.83. The patient presented contralateral upper limb paresis, especially of the deltoid muscle, in the second week post-injection. The electroneuromyography showed neuromuscular block due to botulinum toxin A. She recovered completely after eight months. A switch between different formulations of botulinum toxin type A should prompt caution when carrying out unit conversions. Distant side effects may appear, including paresis in the contralateral limbs.
- Decompressive Surgery in Cerebral Venous Sinus Thrombosis Due to Vaccine‐Induced Immune Thrombotic ThrombocytopeniaPublication . Krzywicka, K; Aguiar de Sousa, D; Cordonnier, C; Bode, F; Field, T; Michalski, D; Pelz, J; Skjelland, M; Wiedmann, M; Zimmermann, J; Wittstock, M; Zanotti, B; Ciccone, A; Bandettini di Poggio, M; Borhani‐Haghighi, A; Chatterton, S; Aujayeb, A; Devroye, A; Dizonno, V; Geeraerts, T; Giammello, F; Günther, A; Ichaporia, N; Kleinig, T; Kristoffersen, E; Lemmens, R; De Maistre, E; Mirzaasgari, Z; Payen, JF; Putaala, J; Petruzzellis, M; Raposo, N; Sadeghi‐Hokmabadi, E; Schoenenberger, S; Umaiorubahan, M; Sylaja, P; van de Munckhof, A; Sánchez van Kammen, M; Lindgren, E; Jood, K; Scutelnic, A; Heldner, M; Poli, S; Kruip, M; Arauz, A; Conforto, A; Aaron, S; Middeldorp, S; Tatlisumak, T; Arnold, M; Coutinho, J; Ferro, JBackground and purpose: Cerebral venous sinus thrombosis due to vaccine-induced immune thrombotic thrombocytopenia (CVST-VITT) is an adverse drug reaction occurring after severe acute respiratory syndrome coronavirus 2 (SARS-CoV-2) vaccination. CVST-VITT patients often present with large intracerebral haemorrhages and a high proportion undergoes decompressive surgery. Clinical characteristics, therapeutic management and outcomes of CVST-VITT patients who underwent decompressive surgery are described and predictors of in-hospital mortality in these patients are explored. Methods: Data from an ongoing international registry of patients who developed CVST within 28 days of SARS-CoV-2 vaccination, reported between 29 March 2021 and 10 May 2022, were used. Definite, probable and possible VITT cases, as defined by Pavord et al. (N Engl J Med 2021; 385: 1680-1689), were included. Results: Decompressive surgery was performed in 34/128 (27%) patients with CVST-VITT. In-hospital mortality was 22/34 (65%) in the surgical and 27/94 (29%) in the non-surgical group (p < 0.001). In all surgical cases, the cause of death was brain herniation. The highest mortality rates were found amongst patients with preoperative coma (17/18, 94% vs. 4/14, 29% in the non-comatose; p < 0.001) and bilaterally absent pupillary reflexes (7/7, 100% vs. 6/9, 67% with unilaterally reactive pupil, and 4/11, 36% with bilaterally reactive pupils; p = 0.023). Postoperative imaging revealed worsening of index haemorrhagic lesion in 19 (70%) patients and new haemorrhagic lesions in 16 (59%) patients. At a median follow-up of 6 months, 8/10 of surgical CVST-VITT who survived admission were functionally independent. Conclusions: Almost two-thirds of surgical CVST-VITT patients died during hospital admission. Preoperative coma and bilateral absence of pupillary responses were associated with higher mortality rates. Survivors often achieved functional independence.