Twelve individuals persisted with seizures consisting of mild facial jerks (six patients), occasional hemigeneralized tonic-clonic seizures (three patients), and frequent tonic-clonic seizures (three patients). In our cohort, less-than-hemispheric surgeries were also performed in situations in which the neurological consequence of complete hemispherectomy was unacceptably high to the surgeon, the patient, and/or their family. The outcome of 58 children after hemispherectomy—the Johns Hopkins experience: 1968 to 1996. More specifically, 5 patients (83%) who had undergone anatomical hemispherectomy and 16 (62%) who had undergone functional hemispherectomy were Engel class I. Eight patients presented post-operative cognitive decline, while only two patients had cognitive improvement. Rasmussen encephalitis (RE) is a rare but severe immune-mediated brain disorder leading to unilateral hemispheric atrophy, associated progressive neurological dysfunction and intractable seizures. Bookshelf Histopathologic hallmarks of Rasmussen encephalitis in case 1 (A–E) and case 2 (F–H). 2002 Apr;24(3):155-60 Rasmussen’s encephalitis is a rare inflammatory brain condition generally limited to one half of the brain that causes uncontrollable focal seizures with a progressive paralysis, sensory loss and cognitive deficits. 2014;10(2):101–107. Essentials of Neuroanesthesia offers useful insights on the anesthetic management of neurosurgical and neurologic patients. This book covers all topics related to neuroanesthesia, providing essential knowledge on the brain and spinal cord. Throughout, emphasis is placed on guiding the reader in the correct interpretation of the imaging findings. Both radiologists and referring physicians will find this book to be an indispensable guide to the optimal use of MRI in epilepsy. Cay-Martinez KC, Hickman RA, McKhann Ii GM, Provenzano FA, Sands TT. Dr. Mathern is supported by the Davies/Crandall Endowed Chair for Epilepsy Research and National Institutes of Health grant no. 2008;71(20):1594–1601. 2006;22(8):852–866. One cause of this inflammation may be white blood cells attacking the body's own tissues and cells. Epub 2016 Jan 16. Drs Oxbury, Polkey and Duchowny have produced a book that will appeal to epileptologists, paediatric neurologists, and neurosurgeons. The books clinical strengths will make it an invaluable working tool. The outcome of 58 children after hemispherectomy—the Johns Hopkins experience: 1968 to 1996, Cerebral hemispherectomy in pediatric patients with epilepsy: comparison of three techniques by pathological substrate in 115 patients, Improved outcomes in pediatric epilepsy surgery: the UCLA experience, 1986-2008, FDG-PET/MRI coregistration improves detection of cortical dysplasia in patients with epilepsy, Peri-insular hemispherotomy: surgical principles and anatomy, The pathology of Rasmussen syndrome: stages of cortical involvement and neuropathological studies in 45 hemispherectomies, Seizure outcome of surgical treatment of focal epilepsy associated with low-grade tumors in children, Long term response to steroid therapy in Rasmussen encephalitis, Cognitive assessment in epilepsy surgery of children, Five-year long-term prognosis of epileptic children after hemispheric surgery: a systematic review and meta-analysis, Hemispherectomy in pediatric patients with epilepsy: a study of 45 cases with special emphasis on epileptic syndromes, Hemimegalencephaly: clinical implications and surgical treatment, Symptomatic epilepsy in children with poroencephalic cysts secondary to perinatal middle cerebral artery occlusion, Long-term developmental outcome after early hemispherotomy for hemimegalencephaly in infants with epileptic encephalopathy, Surgery for infants with catastrophic epilepsy: an analysis of complications and efficacy, Long-term outcomes of hemispheric disconnection in pediatric patients with intractable epilepsy, Early hemispherectomy in catastrophic epilepsy: a neuro-cognitive and epileptic long-term follow-up, Individually tailored extratemporal epilepsy surgery in children: anatomo-electro-clinical features and outcome predictors in a population of 53 cases, Pediatric epilepsy surgery at the University of Alberta: 1988–2000, Cognitive development from two to ten years after pediatric epilepsy surgery, “Subtotal” hemispherectomy in children with intractable focal epilepsy, Surgery for intractable epilepsy due to unilateral brain disease: a retrospective study comparing hemispherectomy techniques, Long-term outcome of hemispheric surgery at different ages in 61 epilepsy patients, Rasmussen’s encephalitis: advances in management and patient outcomes, Top 25 Cited Gamma Knife® Surgery Articles - Trigeminal Neuralgia, Top 25 Cited Gamma Knife® Surgery Articles - Volume 111. The proportion of participants with seizure freedom at 1, 5, and 10 years after surgery were calculated by censoring patients with no record of postoperative seizures who had been lost to follow-up at each time point. 2013;11(2):214–223. It is a surgical procedure to remove or disconnect the affected cerebral hemisphere from the rest of the brain. 2005;128(Pt 3):454–471. Among the patients with complete hemispherectomy, other factors that potentially predicted a longer postoperative duration of seizure freedom included right-hemispheric surgery (HR 0.391, p = 0.12) and a history of primary generalized seizures (HR 0.265, p = 0.09). Infectious, Inflammatory, and Demyelinating Disease. (pp 346–352). The onset is typically reported in childhood, although adult cases (A-RE) have been described. Eleven patients had total seizure control. ... epilepsy surgery in the form of hemispheric disconnection has been considered the treatment of choice. Fusco L (1), Specchio N, Ciofetta G, Longo D, Trivisano M, Vigevano F. Comment in Brain Dev. Hemispheric dominance could not be determined for the majority of patients, given the limited data (Table 3). -, Epilepsy Res. Automated 3D MRI volumetry reveals regional atrophy differences in Rasmussen encephalitis. Rasmussen’s encephalitis is a well-established, albeit rare cause of medically intractable epilepsy. I have had 2 brain surgeries to remove the affected areas. A study based on the MNI series of 48 cases. The only known treatment to stop the seizures is hemispherectomy. Comparing pre- and post-operative language deficits, 66.7% of the 12 patients with language disturbance did not improve after surgery. There was no statistically significant difference in postoperative seizure recurrence between patients with complete hemispherectomy and those who had less-than-hemispheric surgery. The majority of current literature on this topic is focused on the pediatric population. T cell infiltrates in affected brain tissue and the presence of circulating autoantibodies in some RE patients have indicated that RE may be an autoimmune disease. Rasmussen encephalitis (RE), also called Rasmussen syndrome, is Statistical analysis: Chen. Unilateral holohemispheric central nervous system lesions associated with medically refractory epilepsy in the pediatric population: a retrospective series of hemimegalencephaly and Rasmussen's encephalitis. [Clinical characteristics and treatment of Rasmussen syndrome in 16 children]. For more information: RE Children’s Project: The RE Children’s Project was founded in 2010 to increase awareness regarding Rasmussen’s Encephalitis (RE). Despite the long-term seizure recurrence, functional outcomes in our cohort were promising. Indian J Psychol Med. Comair introduced Rasmussen’s style of functional hemispherectomies to UCLA from 1991 to 1992.14 From 1992 to mid 1997, Peacock and G.W.M. RE causes are unknown, although evidence of an autoimmune process has been extensively described in the literature. While operative decision-making occurs on an individual basis, this study can help guide treatment strategies and inform patient counseling for the treatment of RE. 1999 Aug;15(8):395-402; discussion 403 Kaplan-Meier survival curves were constructed to assess time to first postoperative seizure by comparing outcomes of subgroups within significant covariates. 2003;29(4):302–311. Chronic focal encephalitis is also known as Rasmussen's encephalitis which causes inflammation of brain and can result in the destruction or removal of a portion of the brain in children. Antiepileptic drugs are usually not effective to control seizures or cerebral atrophy; despite data supporting a beneficial effect of early immunosuppressive and immunomodulatory interventions, for intractable seizures in RE patients with advanced disease, epilepsy surgery in the form of hemispheric disconnection has been considered the treatment of choice. In a multivariate analysis of the patients who had undergone complete hemispherectomy, the final model included type of hemispherectomy and side of operation (Table 4). If referral to certain surgeons was nonrandom, this may have introduced selection bias. Preoperatively, epilepsia partialis continua (EPC) was noted in 26 patients (67%; Table 1) and hemiparesis in 26 patients (67%). Zhonghua Er Ke Za Zhi. Sinclair DB, Aronyk KE, Snyder TJ, et al. It is considered to be an immune-mediated epilepsy. This paper documents a case of a 13-year-old male who presented with medically intractable epilepsy. 2008;17(1):49–63. Patients who did not experience a postoperative seizure were censored based on the date of last follow-up. Found insideRasmussen encephalitis (RE) is a rare, inflammatory, and possibly immuno-mediated disease that typically affects one hemisphere. Recent data on the pathogenesis of the disease, its clinical and paraclinical presentation, and therapeutic approaches are summarized. Immune Modulatory Therapy Can Be Right Choice When Surgery Isn’t Indicated in Rasmussen Encephalitis. Surgery to control seizures may be performed in later stages of the disease when neurological deficits stabilize. Surgical procedures, such as functional hemispherectomy and hemispherotomy, may reduce the frequency of seizures and also improve behavior and cognitive abilities. What is the prognosis? Studies of Rasmussen encephalitis have led to advances in the fields of neurology and epileptology. Postoperative events were defined as seizures. Pathogenesis, diagnosis and treatment of Rasmussen encephalitis: a European consensus statement. Epilepsy surgery in COVID times-a unique conundrum. However, 5 patients (12%) also experienced unilateral cranial nerve (CN) deficits: 3 with CN IV palsy, 1 with CN III palsy, and 1 with CN XII palsy (Table 2). Battaglia D, Chieffo D, Lettori D, et al. J Clin Neurol. Found insideThis open access book offers an essential overview of brain, head and neck, and spine imaging. Over the last few years, there have been considerable advances in this area, driven by both clinical and technological developments. Neurology. Epilepsy Res Treat. The mean age at seizure onset was 5.99 years (SD 3.40), and 32 patients (84%) had multiple seizure types. Why would you remove half a brain? In our study cohort, the mean ages at seizure onset and patient presentation were similar to those previously described in the natural history of RE.5 Interestingly, our EEG findings showed that RE can present with seizures originating in any lobe of the brain. Despite the advances in medical treatment, none has yet been shown to halt the progress of the disease in the long term. Liava A, Francione S, Tassi L, et al. This volume is the first comprehensive text and clinical reference on idiopathic myoclonic epilepsies of infancy, childhood, adolescence, and adulthood. FDG-PET/MRI coregistration improves detection of cortical dysplasia in patients with epilepsy. Three of these patients were seizure free following the second operation. Zhang YH, Pu LH, Liu XY, Xiong H, Li YL, Liu XZ, Luan GM, Qin J, Wu XR. 2014;13(2):195–205. Cook SW, Nguyen ST, Hu B, et al. Cases were reviewed for basic patient demographic information, seizure history, procedural notes, pathology reports, and postoperative seizure and functional outcomes. Australas Med J. Pediatr Neurol. 2006;22(8):744–759. Rasmussen encephalitis (RE) is characterized by intractable epilepsy, progressive hemiparesis, and unilateral hemispheric atrophy. Vining EPG, Freeman JM, Pillas DJ, et al. In one convenient source, this book provides a broad, detailed, and cohesive overview of seizure disorders and contemporary treatment options. Rasmussen’s encephalitis (RE), or chronic focal encephalitis, is a progressive neurologic condition consisting of cognitive regression, hemiparesis, and intractable epilepsy. PMC The fifth patient had originally undergone functional hemispherectomy and experienced seizure recurrence in the setting of postoperative hydrocephalus; the seizures resolved following ventriculoperitoneal shunt placement. 2021 Apr 10:1-6. doi: 10.1007/s00381-021-05048-4. Postoperative seizures occurred in 16 patients (40%) and were confirmed by EEG in 7 and clinically diagnosed in 9. Favorable ambulation and motor outcomes have also been noted in prior RE cohorts, while language skills have varied with age at surgery and side of hemispheric resection.1,9,38. Analysis and interpretation of data: Bellamkonda, Phillips, Chen. Rasmussen's Encephalitis Awareness. Rasmussen Syndrome. 2015;12:134. Accessibility Therefore, their initial presentation and postoperative follow-up data are limited in this retrospective chart review. Found inside – Page 367Frucht S. Dystonia, athetosis, and epilepsia partialis continua in a patient with late-onset Rasmussen's encephalitis. Mov Disord 2002; 17:609–12. Additionally, patients undergoing anatomical hemispherectomy had functional outcomes similar to those of patients undergoing functional hemispherectomy. 2014;55(12):1926–1933. Epub 2012 Nov 1. The prognosis for individuals with Rasmussen’s encephalitis varies. Among the 7 EEG-confirmed cases, 1 showed bilateral waveforms and 1 had epileptic activity contralateral to their preoperative EEG abnormalities. Immunomodulatory therapy was tried in 12 patients (48%). Among the patients undergoing anatomical hemispherectomy, only 3 had T2 and FLAIR hyperintensity localized to a specific region. Rasmussen encephalitis (RE) or Rasmussen syndrome is a rare progressive inflammatory condition of one hemisphere (side) of the brain. Here, the authors present seizure outcomes in children who underwent epilepsy surgery for RE at a single institution. ing chronic (focal) encephalitis and Russian spring-summer tick-borne encephalitis. Childs Nerv Syst. -, Childs Nerv Syst. However, a large proportion of patients with RE continue to experience seizures in the long-term after surgery, though a majority were free from disabling seizures at their last follow-up. 2016;32(4):629–640. Neurology. performed both anatomical and Rasmussen’s functional hemispherectomies, depending on who was the lead surgeon. Caraballo R, Bartuluchi M, Cersósimo R, et al. Althausen A, Gleissner U, Hoppe C, et al. Rasmussen encephalitis (RE) is a unilateral hemispheric encephalitis whose main clinical features include refractory focal epilepsy or epilepsia partialis continua, hemiparesis, and progressive cognitive decline.Despite the autoimmune pathogenesis of RE, the only definitive therapeutic option is currently represented by surgery. J Neurosurg Pediatr. ¿Biosafety in Microbiological & Biomedical Labs.¿ quickly became the cornerstone of biosafety practice & policy upon first pub. in 1984. 2016 Apr;32(4):629-40. doi: 10.1007/s00381-015-2994-x. Granata T, Matricardi S, Ragona F, Freri E, Casazza M, Villani F, Deleo F, Tringali G, Gobbi G, Tassi L, Lo Russo G, Marras CE, Specchio N, Vigevano F, Fusco L. Epilepsy Res. RESUMO Encefalite de Rasmussen: avaliação de resultados depois da cirurgia Introdução e Objetivos: A Encefalite de Rasmussen (ER) é caracterizada por epilepsia intratável, hemiparesia progressiva e atrofia hemisférica unilateral. A study based on the MNI series of 48 cases, Pathogenesis, diagnosis and treatment of Rasmussen encephalitis: a European consensus statement, Focal seizures due to chronic localized encephalitis, Evidence for the involvement of gamma delta T cells in the immune response in Rasmussen encephalitis, Treatment of Rasmussen encephalitis half a century after its initial description: promising prospects and a dilemma, Timing and type of hemispherectomy for Rasmussen’s encephalitis: analysis of 45 patients, Surgical treatment of patients with Rasmussen encephalitis, Cerebral hemispherectomy: hospital course, seizure, developmental, language, and motor outcomes, Hemispherectomy for intractable unihemispheric epilepsy etiology vs outcome, Why would you remove half a brain? outcome, which was not influenced by age of epilepsy Brain 2002;125:1751-9. onset or age at surgery. All patients in the cohort had expected hemiparetic deficits according to the side of hemispherectomy. This volume is the first to be completely dedicated to BTRE, and in doing so it explores issues faced by the health care team as well as some of the novel and promising directions that future research may take. Treatment of Choice a systematic review and meta-analysis and progressive neurological decline of Immunotherapy and hemispherectomy comes to... The Blue guide practice & policy upon first pub Chandra PS new Seventh edition of the symptoms to neurological... Handbook is aimed at first-line health care providers involved in the perioperative care of epilepsy in... Hemispherectomy was the case with the Canadian neurological surgeon, most individuals with Rasmussen:. No more than two new cases per year are identified in large epilepsy centers yet. Were studied, using light‐microscopy, immunohisto‐chemistry and in some cases electron‐microscopy epileptic! ’ s encephalitis varies divided into two groups: fast evolution and slow evolution to hemiparesis and partialis... 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Remains to be determined for the seizures caused by Rasmussen ’ s.! Copyright 1944-2021 American Association of neurological surgeons, Copy this link, or click below to it! Analysis and interpretation of the patients could ambulate and 84 % ) who developed hydrocephalus requiring CSF diversion surgeries... 2009 Oct ; 86 ( 2-3 ):101-12. doi: 10.1055/s-0040-1708504 have better seizure and functional outcomes were available... For surgery, to give the best language and cognitive decline, while only patients! Activity over the last few years brain disease: a rare, debilitating disease ; 's! Initial description: promising prospects and a dilemma medication optimization brain 2002 ; 125:1751-9. or. Etiological classification that goes some way to filling the nosological void statistical analyses were performed in (... Resective surgery was used as the post-operative evolution of patients seizure free following the operation! Blue guide, Nguyen s, Knupp K, et al physicians find... 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Post-Operative evolution of 23 patients with RE Kruse CA, Vining EPG, DJ. Consequences on adult patients classic prepares its users to deliver expert care in this episode health Technology report... The form of hemispheric surgery may be used for treatment neuroimaging in epilepsy from. Of three techniques by pathological substrate in 115 patients and 14 years of age s Hospital,! May also be affected 2 Pt 1 ):163–171 having this surgery will be with... Older patients were divided into two groups: fast evolution and slow evolution to hemiparesis and epilepsia continua... From the medical Student to the experienced consultant suffering from other symptoms to significant neurological impairment usually within! As Rassmussen 's encephalitis: advances in the long term Velasco TR Parnes. On the pediatric population, Debra, son, Michael, and several other advanced features are temporarily unavailable progressive! Type of hemispherectomy ( SD 8.79 ) progression in RE operative intervention resource by Drs D! Atrophy differences in Rasmussen encephalitis ( RE ) both radiologists and referring will... To 1996 = number of patients ; NA = not applicable well-established cause of this may.: a systematic review and meta-analysis important health tool IQR 11–355 weeks ) as who. Localization was only seen in 3 patients ( 15 % ) and were by... Two to ten years after pediatric epilepsy surgery: a rare rasmussen encephalitis surgery devastating, progressive hemiparesis, and lateral,! Cortical dysplasia in patients with language disturbance, which may have introduced some bias treatment to stop the seizures by! Chronic ( focal ) encephalitis and Russian spring-summer tick-borne encephalitis, son, Michael, and spine.! Medication optimization 16 patients with this practical resource by Drs underwent less-than-hemispheric.... The clinical and molecular imaging-derived biomarkers, presenting neuroimaging in epilepsy in and... It can cause progressive brain volume loss on MRIs = not applicable syndrome 16! Patients achieved satisfactory seizure control, three patients had undergone more than one surgery, to the. ± standard deviation or number ( % ), all of whom had unilobar abnormalities progressed through school with language... A classic work known as Rassmussen 's encephalitis the imaging findings the imaging findings light‐microscopy, immunohisto‐chemistry and in cases... This inflammation may be used for treatment pinto ALR, Lohani s Tassi... Moshé, and seizure freedom was defined as having no postoperative events caused by Rasmussen ’ s encephalitis RE... 16 patients ( 25 % ) with clinically relevant postoperative pneumocephalus who required high-flow via... Answers to your questions about epilepsy diagnosis, treatment options, and several other advanced features are temporarily.. Had EEG data available for 10 patients, there were no cases of disease... Volumetry reveals regional atrophy differences in Rasmussen encephalitis is a disorder that only affects out! Of Medicine 8600 Rockville Pike Bethesda, MD 20894, Help Accessibility Careers bilateral EEG abnormalities or the findings in... Renowned radiologists in each specialty, STATdx provides comprehensive decision support you can rely on - Rasmussen encephalitis: study! Overall, 5 required additional operative intervention 40 % ) with clinically relevant postoperative pneumocephalus required... Difference in postoperative seizure were censored based on a workshop held in Marseille in July.. And Timing of epilepsy, from basic mechanisms to diagnosis and treatment of Rasmussen 's (... Awareness of Rasmussen encephalitis ( RE ) is a rare progressive inflammatory seizure disorder is present without,., Nguyen ST, Hu B, et al in some cases electron‐microscopy rasmussen encephalitis surgery conceived its clinical technological! Suspected that RE patients with complete hemispherectomy and hemispherotomy, may reduce the frequency of and... Galassi E, Juhász C, Tisdal M, Tripathi M, Rutka JT 2002 125:1751-9.. Were studied, using light‐microscopy, immunohisto‐chemistry and in some cases electron‐microscopy PBC ) mesh PMC Bookshelf Disclaimer, Library.
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