Rasmussen’s encephalitis: structural, functional, and clinical correlates of contralesional epileptiform activity

Author:

Bauer TobiasORCID,von Wrede Randi D.,Pujar Suresh,Rácz Attila,Hoppe Christian,Baumgartner Tobias,Varadkar Sophia,Held Nina R.,Reiter Johannes T.,Enders Selma,David Bastian,Prillwitz Conrad C.,Brugues Mar,Keil Vera C. W.,Jeub Monika,Borger Valeri,Sander Josemir W.,Kunz Wolfram S.,Radbruch Alexander,Weber Bernd,Helmstaedter Christoph,Vatter Hartmut,Baldeweg Torsten,Becker Albert J.,Cross J. Helen,Surges Rainer,Rüber TheodorORCID

Abstract

AbstractProgressive inflammation of one hemisphere characterises Rasmussen’s encephalitis (RE), but contralesional epileptiform activity has been repeatedly reported. We aimed to quantify contralesional epileptiform activity in RE and uncover its functional and structural underpinnings. We retrospectively ascertained people with RE treated between 2000 and 2018 at a tertiary centre (Centre 1) and reviewed all available EEG datasets. The temporal occurrence of preoperative contralesional epileptiform activity (interictal/ictal) was evaluated using mixed-effects logistic regression. Cases with/without contralesional epileptiform activity were compared for cognition, inflammation (ipsilesional brain biopsies), and MRI (cortical and fixel-based morphometry). EEG findings were validated in a second cohort treated at another tertiary centre (Centre 2) between 1995 and 2020. We included 127 people with RE and 687 EEG samples. Preoperatively, contralesional epileptiform activity was seen in 30/68 (44%, Centre 1) and 8/59 (14%, Centre 2). In both cohorts, this activity was associated with younger onset age (OR = 0.9; 95% CI 0.83–0.97; P = 0.006). At centre 1, contralesional epileptiform activity was associated with contralesional MRI alterations, lower intelligence (OR = 5.19; 95% CI 1.28–21.08; P = 0.021), and impaired verbal memory (OR = 10.29; 95% CI 1.97–53.85; P = 0.006). After hemispherotomy, 11/17 (65%, Centre 1) and 28/37 (76%, Centre 2) were seizure-free. Contralesional epileptiform activity was persistent postoperatively in 6/12 (50%, Centre 1) and 2/34 (6%, Centre 2). Preoperative contralesional epileptiform activity reduced the chance of postoperative seizure freedom in both cohorts (OR = 0.69; 95% CI 0.50–0.95; P = 0.029). Our findings question the concept of strict unilaterality of RE and provide the evidence of contralesional epileptiform activity as a possible EEG predictor for persisting postoperative seizures.

Funder

BONFOR research commission of the Medical Faculty of the University of Bonn

Universitätsklinikum Bonn

Publisher

Springer Science and Business Media LLC

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