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1. Author Correction: Para-infectious brain injury in COVID-19 persists at follow-up despite attenuated cytokine and autoantibody responses

Author

Michael, Benedict D., Dunai, Cordelia, Needham, Edward J., Tharmaratnam, Kukatharmini, Williams, Robyn, Huang, Yun, Boardman, Sarah A., Clark, Jordan J., Sharma, Parul, Subramaniam, Krishanthi, Wood, Greta K., Collie, Ceryce, Digby, Richard, Ren, Alexander, Norton, Emma, Leibowitz, Maya, Ebrahimi, Soraya, Fower, Andrew, Fox, Hannah, Tato, Esteban, Ellul, Mark A., Sunderland, Geraint, Held, Marie, Hetherington, Claire, Egbe, Franklyn N., Palmos, Alish, Stirrups, Kathy, Grundmann, Alexander, Chiollaz, Anne-Cecile, Sanchez, Jean-Charles, Stewart, James P., Griffiths, Michael, Solomon, Tom, Breen, Gerome, Coles, Alasdair J., Kingston, Nathalie, Bradley, John R., Chinnery, Patrick F., Cavanagh, Jonathan, Irani, Sarosh R., Vincent, Angela, Baillie, J. Kenneth, Openshaw, Peter J., Semple, Malcolm G., Taams, Leonie S., and Menon, David K.

Published
2024
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4. Para-infectious brain injury in COVID-19 persists at follow-up despite attenuated cytokine and autoantibody responses

Author

Michael, Benedict D., Dunai, Cordelia, Needham, Edward J., Tharmaratnam, Kukatharmini, Williams, Robyn, Huang, Yun, Boardman, Sarah A., Clark, Jordan J., Sharma, Parul, Subramaniam, Krishanthi, Wood, Greta K., Collie, Ceryce, Digby, Richard, Ren, Alexander, Norton, Emma, Leibowitz, Maya, Ebrahimi, Soraya, Fower, Andrew, Fox, Hannah, Tato, Esteban, Ellul, Mark A., Sunderland, Geraint, Held, Marie, Hetherington, Claire, Egbe, Franklyn N., Palmos, Alish, Stirrups, Kathy, Grundmann, Alexander, Chiollaz, Anne-Cecile, Sanchez, Jean-Charles, Stewart, James P., Griffiths, Michael, Solomon, Tom, Breen, Gerome, Coles, Alasdair J., Kingston, Nathalie, Bradley, John R., Chinnery, Patrick F., Cavanagh, Jonathan, Irani, Sarosh R., Vincent, Angela, Baillie, J. Kenneth, Openshaw, Peter J., Semple, Malcolm G., Taams, Leonie S., and Menon, David K.

Published
2023
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5. Minimal manifestation status and prednisone withdrawal in the MGTX trial.

Author

Lee, Ikjae, Kuo, Hui-Chien, Aban, Inmaculada B, Cutter, Gary R, McPherson, Tarrant, Kaminski, Henry J, Sussman, Jon, Ströbel, Philipp, Oger, Joel, Cea, Gabriel, Heckmann, Jeannine M, Evoli, Amelia, Nix, Wilfred, Ciafaloni, Emma, Antonini, Giovanni, Witoonpanich, Rawiphan, King, John O, Beydoun, Said R, Chalk, Colin H, Barboi, Alexandru C, Amato, Anthony A, Shaibani, Aziz I, Katirji, Bashar, Lecky, Bryan RF, Buckley, Camilla, Vincent, Angela, Dias-Tosta, Elza, Yoshikawa, Hiroaki, Waddington-Cruz, Marcia, Pulley, Michael T, Rivner, Michael H, Kostera-Pruszczyk, Anna, Pascuzzi, Robert M, Jackson, Carlayne E, Verschuuren, Jan JG, Massey, Janice M, Kissel, John T, Werneck, Lineu C, Benatar, Michael, Barohn, Richard J, Tandan, Rup, Mozaffar, Tahseen, Conwit, Robin, Minisman, Greg, Sonett, Joshua R, and Wolfe, Gil I

Subjects
Biomedical and Clinical Sciences, Clinical Sciences, Clinical Research, Clinical Trials and Supportive Activities, Myasthenia Gravis, Autoimmune Disease, Neurosciences, Rare Diseases, Evaluation of treatments and therapeutic interventions, 6.1 Pharmaceuticals, Adolescent, Adult, Animals, Combined Modality Therapy, Female, Humans, Immunosuppressive Agents, Male, Middle Aged, Prednisone, Rats, Single-Blind Method, Substance Withdrawal Syndrome, Thymectomy, Thymoma, Thymus Neoplasms, Young Adult, MGTX study group, Cognitive Sciences, Neurology & Neurosurgery, Clinical sciences
Abstract

ObjectiveTo examine whether sustained minimal manifestation status (MMS) with complete withdrawal of prednisone is better achieved in thymectomized patients with myasthenia gravis (MG).MethodsThis study is a post hoc analysis of data from a randomized trial of thymectomy in MG (Thymectomy Trial in Non-Thymomatous Myasthenia Gravis Patients Receiving Prednisone Therapy [MGTX]). MGTX was a multicenter, randomized, rater-blinded 3-year trial that was followed by a voluntary 2-year extension for patients with acetylcholine receptor (AChR) antibody-positive MG without thymoma. Patients were randomized 1:1 to thymectomy plus prednisone vs prednisone alone. Participants were age 18-65 years at enrollment with disease duration less than 5 years. All patients received oral prednisone titrated up to 100 mg on alternate days until they achieved MMS, which prompted a standardized prednisone taper as long as MMS was maintained. The achievement rate of sustained MMS (no symptoms of MG for 6 months) with complete withdrawal of prednisone was compared between the thymectomy plus prednisone and prednisone alone groups.ResultsPatients with MG in the thymectomy plus prednisone group achieved sustained MMS with complete withdrawal of prednisone more frequently (64% vs 38%) and quickly compared to the prednisone alone group (median time 30 months vs no median time achieved, p < 0.001) over the 5-year study period. Prednisone-associated adverse symptoms were more frequent in the prednisone alone group and distress level increased with higher doses of prednisone.ConclusionsThymectomy benefits patients with MG by increasing the likelihood of achieving sustained MMS with complete withdrawal of prednisone.Clinicaltrialsgov identifierNCT00294658.Classification of evidenceThis study provides Class II evidence that for patients with generalized MG with AChR antibody, those receiving thymectomy plus prednisone are more likely to attain sustained MMS and complete prednisone withdrawal than those on prednisone alone.

Published
2020

6. Significance of neuronal autoantibodies in comparison to infectious etiologies among patients presenting with encephalitis in a region with a high prevalence of infections

Author

Chang, Thashi, Moloney, Teresa, Jacobson, Leslie, Malavige, Neelika, Lohitharajah, Janarthani, Wanigasinghe, Jithangi, Peach, Sian, Woodhall, Mark, Berretta, Antonio, Waters, Patrick, and Vincent, Angela

Subjects
Encephalitis -- Diagnosis -- Causes of -- Distribution, Autoantibodies -- Health aspects, Company distribution practices, Health
Abstract

Byline: Thashi. Chang, Teresa. Moloney, Leslie. Jacobson, Neelika. Malavige, Janarthani. Lohitharajah, Jithangi. Wanigasinghe, Sian. Peach, Mark. Woodhall, Antonio. Berretta, Patrick. Waters, Angela. Vincent Background: Prevalence of antibody-mediated autoimmune encephalitis (AE) [...]

Published
2022

9. Long-term effect of thymectomy plus prednisone versus prednisone alone in patients with non-thymomatous myasthenia gravis: 2-year extension of the MGTX randomised trial

Author

Wolfe, Gil I, Kaminski, Henry J, Aban, Inmaculada B, Minisman, Greg, Kuo, Hui-Chien, Marx, Alexander, Ströbel, Philipp, Mazia, Claudio, Oger, Joel, Cea, J Gabriel, Heckmann, Jeannine M, Evoli, Amelia, Nix, Wilfred, Ciafaloni, Emma, Antonini, Giovanni, Witoonpanich, Rawiphan, King, John O, Beydoun, Said R, Chalk, Colin H, Barboi, Alexandru C, Amato, Anthony A, Shaibani, Aziz I, Katirji, Bashar, Lecky, Bryan RF, Buckley, Camilla, Vincent, Angela, Dias-Tosta, Elza, Yoshikawa, Hiroaki, Waddington-Cruz, Márcia, Pulley, Michael T, Rivner, Michael H, Kostera-Pruszczyk, Anna, Pascuzzi, Robert M, Jackson, Carlayne E, Verschuuren, Jan JGM, Massey, Janice M, Kissel, John T, Werneck, Lineu C, Benatar, Michael, Barohn, Richard J, Tandan, Rup, Mozaffar, Tahseen, Silvestri, Nicholas J, Conwit, Robin, Sonett, Joshua R, Jaretzki, Alfred, Newsom-Davis, John, Cutter, Gary R, Group, MGTX Study, Cutter, Gary, Aban, Inmaculada, Feese, Michelle, Wolfe, Gil, Kaminski, Henry, Sonett, Joshua, Saluto, Valeria, Rosenberg, Moises, Alvarez, Valeria, Rey, Lisa, King, John, Butzkueven, Helmut, Goldblatt, John, Carey, John, Pollard, John, Reddel, Stephen, Handel, Nicholas, McCaughan, Brian, Pallot, Linda, Novis, Ricardo, Boasquevisque, Carlos, Morato-Fernandez, Rubens, Ximenes, Manoel, Werneck, Lineu, Scola, Rosana, Soltoski, Paulo, Chalk, Colin, Moore, Fraser, Mulder, David, Wadup, Lisa, Mezei, Michele, Evans, Kenneth, Jiwa, Theresa, Schaffar, Anne, White, Chris, Toth, Cory, Gelfand, Gary, Wood, Susan, Pringle, Elizabeth, Zwicker, Jocelyn, Maziak, Donna, Shamji, Farid, and Sundaresan, Sudhir

Subjects
Biomedical and Clinical Sciences, Clinical Sciences, Neurosciences, Myasthenia Gravis, Autoimmune Disease, Rare Diseases, Clinical Research, Clinical Trials and Supportive Activities, Evaluation of treatments and therapeutic interventions, 6.1 Pharmaceuticals, Adult, Female, Humans, Longitudinal Studies, Male, Prednisone, Thymectomy, Treatment Outcome, Young Adult, MGTX Study Group, Neurology & Neurosurgery, Clinical sciences
Abstract

BackgroundThe Thymectomy Trial in Non-Thymomatous Myasthenia Gravis Patients Receiving Prednisone (MGTX) showed that thymectomy combined with prednisone was superior to prednisone alone in improving clinical status as measured by the Quantitative Myasthenia Gravis (QMG) score in patients with generalised non-thymomatous myasthenia gravis at 3 years. We investigated the long-term effects of thymectomy up to 5 years on clinical status, medication requirements, and adverse events.MethodsWe did a rater-blinded 2-year extension study at 36 centres in 15 countries for all patients who completed the randomised controlled MGTX and were willing to participate. MGTX patients were aged 18 to 65 years at enrolment, had generalised non-thymomatous myasthenia gravis of less than 5 years' duration, had acetylcholine receptor antibody titres of 1·00 nmol/L or higher (or concentrations of 0·50-0·99 nmol/L if diagnosis was confirmed by positive edrophonium or abnormal repetitive nerve stimulation, or abnormal single fibre electromyography), had Myasthenia Gravis Foundation of America Clinical Classification Class II-IV disease, and were on optimal anticholinesterase therapy with or without oral corticosteroids. In MGTX, patients were randomly assigned (1:1) to either thymectomy plus prednisone or prednisone alone. All patients in both groups received oral prednisone at doses titrated up to 100 mg on alternate days until they achieved minimal manifestation status. The primary endpoints of the extension phase were the time-weighted means of the QMG score and alternate-day prednisone dose from month 0 to month 60. Analyses were by intention to treat. The trial is registered with ClinicalTrials.gov, number NCT00294658. It is closed to new participants, with follow-up completed.FindingsOf the 111 patients who completed the 3-year MGTX, 68 (61%) entered the extension study between Sept 1, 2009, and Aug 26, 2015 (33 in the prednisone alone group and 35 in the prednisone plus thymectomy group). 50 (74%) patients completed the 60-month assessment, 24 in the prednisone alone group and 26 in the prednisone plus thymectomy group. At 5 years, patients in the thymectomy plus prednisone group had significantly lower time-weighted mean QMG scores (5·47 [SD 3·87] vs 9·34 [5·08]; p=0·0007) and mean alternate-day prednisone doses (24 mg [SD 21] vs 48 mg [29]; p=0·0002) than did those in the prednisone alone group. 14 (42%) of 33 patients in the prednisone group, and 12 (34%) of 35 in the thymectomy plus prednisone group, had at least one adverse event by month 60. No treatment-related deaths were reported during the extension phase.InterpretationAt 5 years, thymectomy plus prednisone continues to confer benefits in patients with generalised non-thymomatous myasthenia gravis compared with prednisone alone. Although caution is appropriate when generalising our findings because of the small sample size of our study, they nevertheless provide further support for the benefits of thymectomy in patients with generalised non-thymomatous myasthenia gravis.FundingNational Institutes of Health, National Institute of Neurological Disorders and Stroke.

Published
2019

11. The importance of early immunotherapy in patients with faciobrachial dystonic seizures

Author

Thompson, Julia, Bi, Mian, Murchison, Andrew G, Makuch, Mateusz, Bien, Christian G, Chu, Kon, Farooque, Pue, Gelfand, Jeffrey M, Geschwind, Michael D, Hirsch, Lawrence J, Somerville, Ernest, Lang, Bethan, Vincent, Angela, Leite, Maria I, Waters, Patrick, Irani, Sarosh R, Dogan-Onugoren, Müjgan, Rae-Grant, Alexander, Illes, Zsolt, Szots, Monika, Malter, Michael, Widman, Guido, Surges, Rainer, Archibald, Neil, Reid, John, Duncan, Callum, Richardson, Anna, Lilleker, James, Iorio, Rafaelle, Blaabjerg, Morten, Abeler, Karin, and Shin, Y

Subjects
Brain Disorders, Neurosciences, Prevention, Clinical Research, Epilepsy, Neurodegenerative, Rare Diseases, ADAM Proteins, Adult, Aged, Aged, 80 and over, Analysis of Variance, Antibodies, Anticonvulsants, Cognition Disorders, Disabled Persons, Female, Flow Cytometry, Follow-Up Studies, Green Fluorescent Proteins, HEK293 Cells, Humans, Immunotherapy, Intracellular Signaling Peptides and Proteins, Limbic Encephalitis, Male, Middle Aged, Nerve Tissue Proteins, Protein Transport, Proteins, Retrospective Studies, Seizures, Surveys and Questionnaires, Transfection, Young Adult, faciobrachial dystonic seizures, leucine-rich glioma-inactivated 1, seizures, immunotherapy, cognitive impairment, Faciobrachial Dystonic Seizures Study Group, Medical and Health Sciences, Psychology and Cognitive Sciences, Neurology & Neurosurgery
Abstract

Faciobrachial dystonic seizures and limbic encephalitis closely associate with antibodies to leucine-rich glioma-inactivated 1 (LGI1). Here, we describe 103 consecutive patients with faciobrachial dystonic seizures and LGI1 antibodies to understand clinical, therapeutic and serological differences between those with and without cognitive impairment, and to determine whether cessation of faciobrachial dystonic seizures can prevent cognitive impairment. The 22/103 patients without cognitive impairment typically had normal brain MRI, EEGs and serum sodium levels (P < 0.0001). Overall, cessation of faciobrachial dystonic seizures with antiepileptic drugs alone occurred in only 9/89 (10%) patients. By contrast, 51% showed cessation of faciobrachial dystonic seizures 30 days after addition of immunotherapy (P < 0.0001), with earlier cessation in cognitively normal patients (P = 0.038). Indeed, expedited immunotherapy (P = 0.031) and normal cognition (P = 0.0014) also predicted reduced disability at 24 months. Furthermore, of 80 patients with faciobrachial dystonic seizures as their initial feature, 56% developed cognitive impairment after 90 days of active faciobrachial dystonic seizures. Whereas only one patient developed cognitive impairment after cessation of faciobrachial dystonic seizures (P < 0.0001). All patients had IgG4-LGI1 antibodies, but those with cognitive impairment had higher proportions of complement-fixing IgG1 antibodies (P = 0.03). Both subclasses caused LGI1-ADAM22 complex internalization, a potential non-inflammatory epileptogenic mechanism. In summary, faciobrachial dystonic seizures show striking time-sensitive responses to immunotherapy, and their cessation can prevent the development of cognitive impairment.awx323media15681705685001.

Published
2018

17. Autoimmune psychosis: an international consensus on an approach to the diagnosis and management of psychosis of suspected autoimmune origin

Author

Pollak, Thomas A, Lennox, Belinda R, Müller, Sabine, Benros, Michael E, Prüss, Harald, Tebartz van Elst, Ludger, Klein, Hans, Steiner, Johann, Frodl, Thomas, Bogerts, Bernhard, Tian, Li, Groc, Laurent, Hasan, Alkomiet, Baune, Bernhard T, Endres, Dominique, Haroon, Ebrahim, Yolken, Robert, Benedetti, Francesco, Halaris, Angelos, Meyer, Jeffrey H, Stassen, Hans, Leboyer, Marion, Fuchs, Dietmar, Otto, Markus, Brown, David A, Vincent, Angela, Najjar, Souhel, and Bechter, Karl

Published
2020
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18. Randomized Trial of Thymectomy in Myasthenia Gravis

Author

Wolfe, Gil I, Kaminski, Henry J, Aban, Inmaculada B, Minisman, Greg, Kuo, Hui-Chien, Marx, Alexander, Ströbel, Philipp, Mazia, Claudio, Oger, Joel, Cea, J Gabriel, Heckmann, Jeannine M, Evoli, Amelia, Nix, Wilfred, Ciafaloni, Emma, Antonini, Giovanni, Witoonpanich, Rawiphan, King, John O, Beydoun, Said R, Chalk, Colin H, Barboi, Alexandru C, Amato, Anthony A, Shaibani, Aziz I, Katirji, Bashar, Lecky, Bryan RF, Buckley, Camilla, Vincent, Angela, Dias-Tosta, Elza, Yoshikawa, Hiroaki, Waddington-Cruz, Márcia, Pulley, Michael T, Rivner, Michael H, Kostera-Pruszczyk, Anna, Pascuzzi, Robert M, Jackson, Carlayne E, Garcia Ramos, Guillermo S, Verschuuren, Jan JGM, Massey, Janice M, Kissel, John T, Werneck, Lineu C, Benatar, Michael, Barohn, Richard J, Tandan, Rup, Mozaffar, Tahseen, Conwit, Robin, Odenkirchen, Joanne, Sonett, Joshua R, Jaretzki, Alfred, Newsom-Davis, John, and Cutter, Gary R

Subjects
Biomedical and Clinical Sciences, Clinical Sciences, Myasthenia Gravis, Clinical Trials and Supportive Activities, Neurosciences, Autoimmune Disease, Clinical Research, Rare Diseases, Evaluation of treatments and therapeutic interventions, 6.1 Pharmaceuticals, Adolescent, Adult, Aged, Combined Modality Therapy, Female, Glucocorticoids, Hospitalization, Humans, Male, Middle Aged, Prednisone, Severity of Illness Index, Single-Blind Method, Thymectomy, Treatment Outcome, Young Adult, MGTX Study Group, Medical and Health Sciences, General & Internal Medicine, Biomedical and clinical sciences, Health sciences
Abstract

BackgroundThymectomy has been a mainstay in the treatment of myasthenia gravis, but there is no conclusive evidence of its benefit. We conducted a multicenter, randomized trial comparing thymectomy plus prednisone with prednisone alone.MethodsWe compared extended transsternal thymectomy plus alternate-day prednisone with alternate-day prednisone alone. Patients 18 to 65 years of age who had generalized nonthymomatous myasthenia gravis with a disease duration of less than 5 years were included if they had Myasthenia Gravis Foundation of America clinical class II to IV disease (on a scale from I to V, with higher classes indicating more severe disease) and elevated circulating concentrations of acetylcholine-receptor antibody. The primary outcomes were the time-weighted average Quantitative Myasthenia Gravis score (on a scale from 0 to 39, with higher scores indicating more severe disease) over a 3-year period, as assessed by means of blinded rating, and the time-weighted average required dose of prednisone over a 3-year period.ResultsA total of 126 patients underwent randomization between 2006 and 2012 at 36 sites. Patients who underwent thymectomy had a lower time-weighted average Quantitative Myasthenia Gravis score over a 3-year period than those who received prednisone alone (6.15 vs. 8.99, P

Published
2016

19. A clinical approach to diagnosis of autoimmune encephalitis

Author

Graus, Francesc, Titulaer, Maarten J, Balu, Ramani, Benseler, Susanne, Bien, Christian G, Cellucci, Tania, Cortese, Irene, Dale, Russell C, Gelfand, Jeffrey M, Geschwind, Michael, Glaser, Carol A, Honnorat, Jerome, Höftberger, Romana, Iizuka, Takahiro, Irani, Sarosh R, Lancaster, Eric, Leypoldt, Frank, Prüss, Harald, Rae-Grant, Alexander, Reindl, Markus, Rosenfeld, Myrna R, Rostásy, Kevin, Saiz, Albert, Venkatesan, Arun, Vincent, Angela, Wandinger, Klaus-Peter, Waters, Patrick, and Dalmau, Josep

Subjects
Biomedical and Clinical Sciences, Clinical Sciences, Immunization, Prevention, Clinical Research, Autoimmune Disease, Brain Disorders, Vaccine Related, Neurosciences, Neurodegenerative, 4.1 Discovery and preclinical testing of markers and technologies, 4.2 Evaluation of markers and technologies, Detection, screening and diagnosis, Metabolic and endocrine, Inflammatory and immune system, Autoimmune Diseases of the Nervous System, Encephalitis, Humans, Neurology & Neurosurgery, Clinical sciences
Abstract

Encephalitis is a severe inflammatory disorder of the brain with many possible causes and a complex differential diagnosis. Advances in autoimmune encephalitis research in the past 10 years have led to the identification of new syndromes and biomarkers that have transformed the diagnostic approach to these disorders. However, existing criteria for autoimmune encephalitis are too reliant on antibody testing and response to immunotherapy, which might delay the diagnosis. We reviewed the literature and gathered the experience of a team of experts with the aims of developing a practical, syndrome-based diagnostic approach to autoimmune encephalitis and providing guidelines to navigate through the differential diagnosis. Because autoantibody test results and response to therapy are not available at disease onset, we based the initial diagnostic approach on neurological assessment and conventional tests that are accessible to most clinicians. Through logical differential diagnosis, levels of evidence for autoimmune encephalitis (possible, probable, or definite) are achieved, which can lead to prompt immunotherapy.

Published
2016

22. Multimodal electrophysiological analyses reveal that reduced synaptic excitatory neurotransmission underlies seizures in a model of NMDAR antibody-mediated encephalitis

Author

Wright, Sukhvir K., Rosch, Richard E., Wilson, Max A., Upadhya, Manoj A., Dhangar, Divya R., Clarke-Bland, Charlie, Wahid, Tamara T., Barman, Sumanta, Goebels, Norbert, Kreye, Jakob, Prüss, Harald, Jacobson, Leslie, Bassett, Danielle S., Vincent, Angela, Greenhill, Stuart D., and Woodhall, Gavin L.

Published
2021
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23. The clinical profile of NMOSD in Australia and New Zealand

Author

Bukhari, Wajih, Clarke, Laura, O’Gorman, Cullen, Khalilidehkordi, Elham, Arnett, Simon, Prain, Kerri M., Woodhall, Mark, Silvestrini, Roger, Bundell, Christine S., Ramanathan, Sudarshini, Abernethy, David, Bhuta, Sandeep, Blum, Stefan, Boggild, Mike, Boundy, Karyn, Brew, Bruce J., Brownlee, Wallace, Butzkueven, Helmut, Carroll, William M., Chen, Celia, Coulthard, Alan, Dale, Russell C., Das, Chandi, Dear, Keith, Fabis-Pedrini, Marzena J., Fulcher, David, Gillis, David, Hawke, Simon, Heard, Robert, Henderson, Andrew P. D., Heshmat, Saman, Hodgkinson, Suzanne, Jimenez-Sanchez, Sofia, Kilpatrick, Trevor J., King, John, Kneebone, Chris, Kornberg, Andrew J., Lechner-Scott, Jeannette, Lin, Ming-Wei, Lynch, Christopher, Macdonnell, Richard A. L., Mason, Deborah F., McCombe, Pamela A., Pereira, Jennifer, Pollard, John D., Reddel, Stephen W., Shaw, Cameron, Spies, Judith, Stankovich, James, Sutton, Ian, Vucic, Steve, Walsh, Michael, Wong, Richard C., Yiu, Eppie M., Barnett, Michael H., Kermode, Allan G., Marriott, Mark P., Parratt, John, Slee, Mark, Taylor, Bruce V., Willoughby, Ernest, Wilson, Robert J., Brilot, Fabienne, Vincent, Angela, Waters, Patrick, and Broadley, Simon A.

Published
2020
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25. Long-term effect of thymectomy plus prednisone versus prednisone alone in patients with non-thymomatous myasthenia gravis: 2-year extension of the MGTX randomised trial

Author

Cutter, Gary, Aban, Inmaculada, Minisman, Greg, Feese, Michelle, Kuo, Hui-Chien, Newsom-Davis, John, Wolfe, Gil, Kaminski, Henry, Jaretzki, Alfred, Sonett, Joshua, Mazia, Claudio, Saluto, Valeria, Rosenberg, Moises, Alvarez, Valeria, Rey, Lisa, King, John, Butzkueven, Helmut, Goldblatt, John, Carey, John, Pollard, John, Reddel, Stephen, Handel, Nicholas, McCaughan, Brian, Pallot, Linda, Waddington-Cruz, Márcia, Novis, Ricardo, Boasquevisque, Carlos, Dias-Tosta, Elza, Morato-Fernandez, Rubens, Ximenes, Manoel, Werneck, Lineu, Scola, Rosana, Soltoski, Paulo, Chalk, Colin, Moore, Fraser, Mulder, David, Wadup, Lisa, Oger, Joel, Mezei, Michele, Evans, Kenneth, Jiwa, Theresa, Schaffar, Anne, White, Chris, Toth, Cory, Gelfand, Gary, Wood, Susan, Pringle, Elizabeth, Zwicker, Jocelyn, Maziak, Donna, Shamji, Farid, Sundaresan, Sudhir, Seely, Andrew, Cea, Gabriel, Verduga, Renato, Aguayo, Alberto, Jander, Sebastian, Zickler, Philipp, Klein, Michael, Marx, Alexander, Ströbel, Philipp, Weis, Cleo-Aron, Melms, Arthur, Bischof, Felix, Aebert, Hermann, Ziemer, Gerhard, Nix, Wilfred, Thümler, Björn, Wilhem-Schwenkmezger, Thomas, Mayer, Eckhard, Schalke, Berthold, Pöschel, Peter, Hieber, Gisela, Wiebe, Karsten, Antonini, Giovanni, Clemenzi, Alessandro, Ceschin, Vanessa, Rendina, Erino, Venuta, Federico, Morino, Stefania, Bucci, Elisabetta, Durelli, Luca, Tavella, Alessia, Clerico, Marinella, Contessa, Giulia, Borasio, Piero, Evoli, Amelia, Servidei, Serenella, Granone, Pierluigi, Mantegazza, Renato, Berta, Emilia, Novellino, Lorenzo, Spinelli, Luisa, Motomura, Masakatsu, Matsuo, Hidenori, Nagayasu, Takeshi, Yoshikawa, Hiroaki, Takamori, Masaharu, Oda, Makoto, Matsumoto, Isao, Furukawa, Yutaka, Noto, Daisuke, Motozaki, Yuko, Iwasa, Kazuo, Yanase, Daisuke, Garcia Ramos, Guillermo, Cacho, Bernardo, de la Garza, Lorenzo, Kostera-Pruszczyk, Anne, Lipowska, Marta, Kwiecinski, Hubert, Potulska-Chromik, Anna, Orlowski, Tadeusz, Silva, Ana, Feijo, Marta, Freitas, António, Heckmann, Jeannine, Frost, Andrew, Pan, Edward, Tucker, Lawrence, Rossouw, Johan, Drummond, Fiona, Illa, Isabel, Diaz, Jorge, Leon, Carlos, Yeh, Jiann-Horng, Chiu, Hou-Chang, Hsieh, Yei-San, Witoonpanich, Rawiphan, Tunlayadechanont, Supoch, Attanavanich, Sukasom, Verschuuren, Jan, Straathof, Chiara, Titulaer, Maarten, Versteegh, Michel, Pels, Arda, Krum, Yvonne, Buckley, Camilla, Leite, M. Isabel, Vincent, Angela, Hilton-Jones, David, Ratnatunga, Chandi, Farrugia, Maria, Petty, Richard, Overell, James, Kirk, Alan, Gibson, Andrew, McDermott, Chris, Hopkinson, David, Lecky, Bryan, Watling, David, Marshall, Dot, Saminaden, Sam, Davies, Deborah, Dougan, Charlotte, Sathasivam, Siva, Page, Richard, Sussman, Jon, Ealing, John, Krysiak, Peter, Amato, Anthony, Salajegheh, Mohammad, Jaklitsch, Michael, Roe, Kristen, Ashizawa, Tetsuo, Smith, Robert Glenn, Zwischenberg, Joseph, Stanton, Penny, Barboi, Alexandru, Jaradeh, Safwan, Tisol, William, Gasparri, Mario, Haasler, George, Yellick, Mary, Dennis, Cedric, Barohn, Richard, Pasnoor, Mamatha, Dimachkie, Mazen, McVey, April, Gronseth, Gary, Dick, Arthur, Kramer, Jeffrey, Currence, Melissa, Herbelin, Laura, Belsh, Jerry, Li, George, Langenfeld, John, Mertz, Mary Ann, Benatar, Michael, Harrison, Taylor, Force, Seth, Usher, Sharon, Beydoun, Said, Lin, Frank, DeMeester, Steve, Akhter, Salem, Malekniazi, Ali, Avenido, Gina, Crum, Brian, Milone, Margherita, Cassivi, Stephen, Fisher, Janet, Ciafaloni, Emma, Heatwole, Chad, Watson, Thomas, Hilbert, James, Smirnow, Alexis, Distad, B. Jane, Weiss, Michael, Wood, Douglas, Haug, Joanna, Ernstoff, Raina, Cao, Jingyang, Chmielewski, Gary, Welsh, Robert, Duris, Robin, Gutmann, Laurie, Pawar, Gauri, Graeber, Geoffrey Marc, Altemus, Patricia, Nance, Christopher, Gutmann, Ludwig, Jackson, Carlayne, Grogan, Patrick, Calhoon, John, Kittrell, Pamela, Myers, Deborah, Hayat, Ghazala, Naunheim, Keith, Eller, Susan, Holzemer, Eve, Katirji, Bashar, Alshekhlee, Amer, Robke, Jason, Karlinchak, Brenda, Katz, Jonathan, Miller, Robert, Roan, Ralph, Forshew, Dallas, Kissel, John, Elsheikh, Bakri, Ross, Patrick, Chelnick, Sharon, Lewis, Richard, Acsadi, Agnes, Baciewicz, Frank, Masse, Stacey, Massey, Janice, Juel, Vern, Onaitis, Mark, Lowe, James, Lipscomb, Bernadette, Mozaffar, Tahseen, Thai, Gaby, Milliken, Jeffrey, Martin, Veronica, Karayan, Ronnie, Muley, Suraj, Parry, Gareth, Shumway, Sara, Oh, Shin, Claussen, Gwen, Lu, Liang, Cerfolio, Robert, Young, Angela, Morgan, Marla, Pascuzzi, Robert, Kincaid, John, Kesler, Kenneth, Guingrich, Sandy, Michaels, Angi, Phillips, Lawrence, Burns, Ted, Jones, David, Fischer, Cindy, Pulley, Michael, Berger, Alan, D'Agostino, Harry, Smith, Lisa, Rivner, Michael, Pruitt, Jerry, Landolfo, Kevin, Hillman, Demetric, Shaibani, Aziz, Sermas, Angelo, Ruel, Ross, Ismail, Farah, Sivak, Mark, Goldstein, Martin, Camunas, Jorge, Bratton, Joan, Tandan, Rup, Panitch, Hill, Leavitt, Bruce, Jones, Marilee, Muppidi, Srikanth, Vernino, Steven, Nations, Sharon, Meyer, Dan, Gorham, Nina, Wolfe, Gil I, Kaminski, Henry J, Aban, Inmaculada B, Cea, J Gabriel, Heckmann, Jeannine M, King, John O, Beydoun, Said R, Chalk, Colin H, Barboi, Alexandru C, Amato, Anthony A, Shaibani, Aziz I, Lecky, Bryan R F, Pulley, Michael T, Rivner, Michael H, Kostera-Pruszczyk, Anna, Pascuzzi, Robert M, Jackson, Carlayne E, Verschuuren, Jan J G M, Massey, Janice M, Kissel, John T, Werneck, Lineu C, Barohn, Richard J, Silvestri, Nicholas J, Conwit, Robin, Sonett, Joshua R, Jaretzki, Alfred, III, and Cutter, Gary R

Published
2019
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26. Cell‐surface central nervous system autoantibodies: Clinical relevance and emerging paradigms

Author

Irani, Sarosh R, Gelfand, Jeffrey M, Al‐Diwani, Adam, and Vincent, Angela

Subjects
Neurodegenerative, Brain Disorders, Neurosciences, Immunization, Autoimmune Disease, Mental Health, Prevention, Vaccine Related, Neurological, Autoantibodies, Central Nervous System, Humans, Membrane Proteins, Nervous System Diseases, Clinical Sciences, Neurology & Neurosurgery
Abstract

The recent discovery of several potentially pathogenic autoantibodies has helped identify patients with clinically distinctive central nervous system diseases that appear to benefit from immunotherapy. The associated autoantibodies are directed against the extracellular domains of cell-surface-expressed neuronal or glial proteins such as LGI1, N-methyl-D-aspartate receptor, and aquaporin-4. The original descriptions of the associated clinical syndromes were phenotypically well circumscribed. However, as availability of antibody testing has increased, the range of associated patient phenotypes and demographics has expanded. This in turn has led to the recognition of more immunotherapy-responsive syndromes in patients presenting with cognitive and behavioral problems, seizures, movement disorders, psychiatric features, and demyelinating disease. Although antibody detection remains diagnostically important, clinical recognition of these distinctive syndromes should ensure early and appropriate immunotherapy administration. We review the emerging paradigm of cell-surface-directed antibody-mediated neurological diseases, describe how the associated disease spectrums have broadened since the original descriptions, discuss some of the methodological issues regarding techniques for antibody detection and emphasize considerations surrounding immunotherapy administration. As these disorders continue to reach mainstream neurology and even psychiatry, more cell-surface-directed antibodies will be discovered, and their possible relevance to other more common disease presentations should become more clearly defined.

Published
2014

28. Rasmussens encephalitis: clinical features, pathobiology, and treatment advances.

Author

Varadkar, Sophia, Bien, Christian, Kruse, Carol, Jensen, Frances, Bauer, Jan, Pardo, Carlos, Vincent, Angela, Cross, J, and Mathern, Gary

Subjects
Encephalitis, Humans
Abstract

Rasmussens encephalitis is a rare chronic neurological disorder, characterised by unilateral inflammation of the cerebral cortex, drug-resistant epilepsy, and progressive neurological and cognitive deterioration. Neuropathological and immunological studies support the notion that Rasmussens encephalitis is probably driven by a T-cell response to one or more antigenic epitopes, with potential additional contribution by autoantibodies. Careful analysis of the association between histopathology and clinical presentation suggests that initial damage to the brain is mediated by T cells and microglia, suggesting a window for treatment if Rasmussens encephalitis can be diagnosed early. Advances in neuroimaging suggest that progression of the inflammatory process seen with MRI might be a good biomarker in Rasmussens encephalitis. For many patients, families, and doctors, choosing the right time to move from medical management to surgery is a real therapeutic dilemma. Cerebral hemispherectomy remains the only cure for seizures, but there are inevitable functional compromises. Decisions of whether or when surgery should be undertaken are challenging in the absence of a dense neurological deficit, and vary by institutional experience. Further, the optimum time for surgery, to give the best language and cognitive outcome, is not yet well understood. Immunomodulatory treatments seem to slow rather than halt disease progression in Rasmussens encephalitis, without changing the eventual outcome.

Published
2014

29. Neuronal antibody prevalence in children with seizures < 3 years: a prospective national cohort

Author

Symonds, Joseph D., Moloney, Teresa C., Lang, Bethan, McLellan, Ailsa, O’Regan, Mary E., MacLeod, Stewart, Jollands, Alice, Vincent, Angela, Kirkpatrick, Martin, Brunklaus, Andreas, Shetty, Jayakara, Dorris, Liam, Forbes, Kirsten, Abu-Arafeh, Ishaq, Andrew, Jamie, Brink, Philip, Callaghan, Mary, Cruden, Jamie, Findlay, Christine, Grattan, Rosemary, MacDonnell, Jane, McKnight, Jean, Morrison, Calum A., Nairn, Lesley, Pilley, Elizabeth, Stephen, Elma, Thomsen, Selina, Webb, Alan, Wilson, Margaret, and Zuberi, Sameer M.

Published
2020
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33. Neuroimaging in encephalitis: analysis of imaging findings and interobserver agreement

Author

Ambrose, Helen E., Clewley, Jonathan P., Walsh, Amanda L., Morgan, Dilys, Cunningham, Richard, Zuckerman, Mark, Mutton, Kenneth J., Ward, Katherine N., Lunn, Michael P.T., Irani, Sarosh R., Vincent, Angela, Ford, Craig, Rothwell, Emily, Tong, William, Lin, Jean-Pierre, Ahmed, Javeed, Cubitt, David, Hemingway, Cheryl, Muir, David, Lyall, Hermione, Thompson, Ed, Keir, Geoff, Worthington, Viki, Griffiths, Paul, Bennett, Susan, Kneen, Rachel, Klapper, Paul, Granerod, J., Davies, N.W.S., Mukonoweshuro, W., Mehta, A., Das, K., Lim, M., Solomon, T., Biswas, S., Rosella, L., Brown, D.W.G., and Crowcroft, N.S.

Published
2016
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38. Clinical predictors of encephalitis in UK adults–A multi-centre prospective observational cohort study.

Author

Defres, Sylviane, Tharmaratnam, Kukatharmini, Michael, Benedict D., Ellul, Mark, Davies, Nicholas W. S., Easton, Ava, Griffiths, Michael J., Bhojak, Maneesh, Das, Kumar, Hardwick, Hayley, Cheyne, Chris, Kneen, Rachel, Medina-Lara, Antonieta, Salter, Anne Christine, Beeching, Nicholas J., Carrol, Enitan, Vincent, Angela, Garcia-Finana, Marta, and Solomon, Tom

Subjects
FEVER, ENCEPHALITIS, COHORT analysis, SCIENTIFIC observation, CEREBROSPINAL fluid, AUTOIMMUNE diseases
Abstract

Objectives: Encephalitis, brain inflammation and swelling, most often caused by an infection or the body's immune defences, can have devastating consequences, especially if diagnosed late. We looked for clinical predictors of different types of encephalitis to help clinicians consider earlier treatment. Methods: We conducted a multicentre prospective observational cohort study (ENCEPH-UK) of adults (> 16 years) with suspected encephalitis at 31 UK hospitals. We evaluated clinical features and investigated for infectious and autoimmune causes. Results: 341 patients were enrolled between December 2012 and December 2015 and followed up for 12 months. 233 had encephalitis, of whom 65 (28%) had HSV, 38 (16%) had confirmed or probable autoimmune encephalitis, and 87 (37%) had no cause found. The median time from admission to 1st dose of aciclovir for those with HSV was 14 hours (IQR 5–50); time to 1st dose of immunosuppressant for the autoimmune group was 125 hours (IQR 45–250). Compared to non-HSV encephalitis, patients with HSV more often had fever, lower serum sodium and lacked a rash. Those with probable or confirmed autoimmune encephalitis were more likely to be female, have abnormal movements, normal serum sodium levels and a cerebrospinal fluid white cell count < 20 cells x106/L, but they were less likely to have a febrile illness. Conclusions: Initiation of treatment for autoimmune encephalitis is delayed considerably compared with HSV encephalitis. Clinical features can help identify patients with autoimmune disease and could be used to initiate earlier presumptive therapy. [ABSTRACT FROM AUTHOR]

Published
2023
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