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51. Randomized trial of three IVIg doses for treating chronic inflammatory demyelinating polyneuropathy

Author

Cornblath, David R., Van Doorn, Pieter A., Hartung, Hans Peter, Merkies, Ingemar S.J., Katzberg, Hans D., Hinterberger, Doris, Clodi, Elisabeth, Cornblath, David R., Van Doorn, Pieter A., Hartung, Hans Peter, Merkies, Ingemar S.J., Katzberg, Hans D., Hinterberger, Doris, and Clodi, Elisabeth

Abstract

Intravenous immunoglobulin treatment for chronic inflammatory demyelinating polyneuropathy usually starts with a 2.0 g/kg induction dose followed by 1.0 g/kg maintenance doses every 3 weeks. No dose-ranging studies with intravenous immunoglobulin maintenance therapy have been published. The Progress in Chronic Inflammatory Demyelinating polyneuropathy (ProCID) study was a prospective, double-blind, randomized, parallel-group, multicentre, phase III study investigating the efficacy and safety of 10% liquid intravenous immunoglobulin (Panzyga®) in patients with active chronic inflammatory demyelinating polyneuropathy. Patients were randomized 1:2:1 to receive the standard intravenous immunoglobulin induction dose and then either 0.5, 1.0 or 2.0 g/kg maintenance doses every 3 weeks. The primary end point was the response rate in the 1.0 g/kg group, defined as an improvement ≥1 point in adjusted Inflammatory Neuropathy Cause and Treatment score at Week 6 versus baseline and maintained at Week 24. Secondary end points included dose response and safety. This trial was registered with EudraCT (Number 2015-005443-14) and clinicaltrials.gov (NCT02638207). Between August 2017 and September 2019, the study enrolled 142 patients. All 142 were included in the safety analyses. As no post-infusion data were available for three patients, 139 were included in the efficacy analyses, of whom 121 were previously on corticosteroids. The response rate was 80% (55/69 patients) [95% confidence interval (CI): 69-88%] in the 1.0 g/kg group, 65% (22/34; CI: 48-79%) in the 0.5 g/kg group, and 92% (33/36; CI: 78-97%) in the 2.0 g/kg group. While the proportion of responders was higher with higher maintenance doses, logistic regression analysis showed that the effect on response rate was driven by a significant difference between the 0.5 and 2.0 g/kg groups, whereas the response rates in the 0.5 and 2.0 g/kg groups did not differ significantly from the 1.0 g/kg group. Fifty-six per cent of al

Published
2022

52. Electrodiagnostic subtyping in Guillain-Barré syndrome:Use of criteria in practice based on a survey study in IGOS

Author

Arends, Samuel, Drenthen, Judith, Van den Bergh, Peter Y.K., Hadden, Robert D.M., Shahrizaila, Nortina, Dimachkie, Mazen M., Gutiérrez Gutiérrez, Gerardo, Katzberg, Hans, Kiers, Lynette, Lehmann, Helmar C., Péréon, Yann, Reisin, Ricardo C., Uncini, Antonino, Verhamme, Camiel, Waheed, Wagar, Cornblath, David R., Jacobs, Bart C., Arends, Samuel, Drenthen, Judith, Van den Bergh, Peter Y.K., Hadden, Robert D.M., Shahrizaila, Nortina, Dimachkie, Mazen M., Gutiérrez Gutiérrez, Gerardo, Katzberg, Hans, Kiers, Lynette, Lehmann, Helmar C., Péréon, Yann, Reisin, Ricardo C., Uncini, Antonino, Verhamme, Camiel, Waheed, Wagar, Cornblath, David R., and Jacobs, Bart C.

Abstract

Electrodiagnostic (EDx) studies are helpful in diagnosing and subtyping of Guillain-Barré syndrome (GBS). Published criteria for differentiation into GBS subtypes focus on cutoff values, but other items receive less attention, although they may influence EDx subtyping: (a) extensiveness of EDx testing, (b) nerve-specific considerations, (c) distal compound muscle action potential (CMAP)-amplitude requirements, (d) criteria for conduction block and temporal dispersion. The aims of this study were to investigate how these aspects were approached by neuromuscular EDx experts in practice and how this was done in previously published EDx criteria for GBS. A completed questionnaire was returned by 24 (of 49) members of the electrophysiology expertise group from the International GBS Outcome Study. Six published EDx criteria for GBS subtyping were compared regarding these aspects. The indicated minimal number of motor nerves to study varied among respondents and tended to be more extensive in equivocal than normal studies. Respondents varied considerably regarding usage of compression sites for subtyping (median/wrist, ulnar/elbow, peroneal/fibular head): 29% used all variables from all sites, 13% excluded all sites, and 58% used only some sites and/or variables. Thirty-eight percent of respondents required a minimal distal CMAP amplitude to classify distal motor latency as demyelinating, and 58% did for motor conduction velocity. For proximal/distal CMAP-amplitude ratio and F-wave latency, a requisite minimal CMAP amplitude was more often required (79%). Also, the various published criteria sets showed differences on all items. Practical use of EDx criteria for subtyping GBS vary extensively across respondents, potentially lowering the reproducibility of GBS subtyping.

Published
2022

53. Predicting Outcome in Guillain-Barré Syndrome: International Validation of the Modified Erasmus GBS Outcome Score

Author

Doets, A, Lingsma, H, Walgaard, C, Islam, B, Papri, N, Davidson, A, Yamagishi, Y, Kusunoki, S, Dimachkie, M, Waheed, W, Kolb, N, Islam, Z, Mohammad, Q, Harbo, T, Sindrup, S, Chavada, G, Willison, H, Casasnovas, C, Bateman, K, Miller, J, van den Berg, B, Verboon, C, Roodbol, J, Leonhard, S, Benedetti, L, Kuwabara, S, Van den Bergh, P, Monges, S, Marfia, G, Shahrizaila, N, Galassi, G, Péréon, Y, Bürmann, J, Kuitwaard, K, Kleyweg, R, Marchesoni, C, Sedano Tous, M, Querol, L, Illa, I, Wang, Y, Nobile-Orazio, E, Rinaldi, S, Schenone, A, Pardo, J, Vermeij, F, Lehmann, H, Granit, V, Cavaletti, G, Gutiérrez-Gutiérrez, G, Barroso, F, Visser, L, Katzberg, H, Dardiotis, E, Attarian, S, van der Kooi, A, Eftimov, F, Wirtz, P, Samijn, J, Gilhuis, H, Hadden, R, Holt, J, Sheikh, K, Karafiath, S, Vytopil, M, Antonini, G, Feasby, T, Faber, C, Gijsbers, C, Busby, M, Roberts, R, Silvestri, N, Fazio, R, van Dijk, G, Garssen, M, Straathof, C, Gorson, K, Jacobs, B, Doets, Alex Y, Lingsma, Hester F, Walgaard, Christa, Islam, Badrul, Papri, Nowshin, Davidson, Amy, Yamagishi, Yuko, Kusunoki, Susumu, Dimachkie, Mazen M, Waheed, Waqar, Kolb, Noah, Islam, Zhahirul, Mohammad, Quazi Deen, Harbo, Thomas, Sindrup, Soren H, Chavada, Govindsinh, Willison, Hugh J, Casasnovas, Carlos, Bateman, Kathleen, Miller, James A L, van den Berg, Bianca, Verboon, Christine, Roodbol, Joyce, Leonhard, Sonja E, Benedetti, Luana, Kuwabara, Satoshi, Van den Bergh, Peter, Monges, Soledad, Marfia, Girolama A, Shahrizaila, Nortina, Galassi, Giuliana, Péréon, Yann, Bürmann, Jan, Kuitwaard, Krista, Kleyweg, Ruud P, Marchesoni, Cintia, Sedano Tous, María J, Querol, Luis, Illa, Isabel, Wang, Yuzhong, Nobile-Orazio, Eduardo, Rinaldi, Simon, Schenone, Angelo, Pardo, Julio, Vermeij, Frederique H, Lehmann, Helmar C, Granit, Volkan, Cavaletti, Guido, Gutiérrez-Gutiérrez, Gerardo, Barroso, Fabio A, Visser, Leo H, Katzberg, Hans D, Dardiotis, Efthimios, Attarian, Shahram, van der Kooi, Anneke J, Eftimov, Filip, Wirtz, Paul W, Samijn, Johnny P A, Gilhuis, H Jacobus, Hadden, Robert D M, Holt, James K L, Sheikh, Kazim A, Karafiath, Summer, Vytopil, Michal, Antonini, Giovanni, Feasby, Thomas E, Faber, Catharina G, Gijsbers, Cees J, Busby, Mark, Roberts, Rhys C, Silvestri, Nicholas J, Fazio, Raffaella, van Dijk, Gert W, Garssen, Marcel P J, Straathof, Chiara S M, Gorson, Kenneth C, Jacobs, Bart C, Doets, A, Lingsma, H, Walgaard, C, Islam, B, Papri, N, Davidson, A, Yamagishi, Y, Kusunoki, S, Dimachkie, M, Waheed, W, Kolb, N, Islam, Z, Mohammad, Q, Harbo, T, Sindrup, S, Chavada, G, Willison, H, Casasnovas, C, Bateman, K, Miller, J, van den Berg, B, Verboon, C, Roodbol, J, Leonhard, S, Benedetti, L, Kuwabara, S, Van den Bergh, P, Monges, S, Marfia, G, Shahrizaila, N, Galassi, G, Péréon, Y, Bürmann, J, Kuitwaard, K, Kleyweg, R, Marchesoni, C, Sedano Tous, M, Querol, L, Illa, I, Wang, Y, Nobile-Orazio, E, Rinaldi, S, Schenone, A, Pardo, J, Vermeij, F, Lehmann, H, Granit, V, Cavaletti, G, Gutiérrez-Gutiérrez, G, Barroso, F, Visser, L, Katzberg, H, Dardiotis, E, Attarian, S, van der Kooi, A, Eftimov, F, Wirtz, P, Samijn, J, Gilhuis, H, Hadden, R, Holt, J, Sheikh, K, Karafiath, S, Vytopil, M, Antonini, G, Feasby, T, Faber, C, Gijsbers, C, Busby, M, Roberts, R, Silvestri, N, Fazio, R, van Dijk, G, Garssen, M, Straathof, C, Gorson, K, Jacobs, B, Doets, Alex Y, Lingsma, Hester F, Walgaard, Christa, Islam, Badrul, Papri, Nowshin, Davidson, Amy, Yamagishi, Yuko, Kusunoki, Susumu, Dimachkie, Mazen M, Waheed, Waqar, Kolb, Noah, Islam, Zhahirul, Mohammad, Quazi Deen, Harbo, Thomas, Sindrup, Soren H, Chavada, Govindsinh, Willison, Hugh J, Casasnovas, Carlos, Bateman, Kathleen, Miller, James A L, van den Berg, Bianca, Verboon, Christine, Roodbol, Joyce, Leonhard, Sonja E, Benedetti, Luana, Kuwabara, Satoshi, Van den Bergh, Peter, Monges, Soledad, Marfia, Girolama A, Shahrizaila, Nortina, Galassi, Giuliana, Péréon, Yann, Bürmann, Jan, Kuitwaard, Krista, Kleyweg, Ruud P, Marchesoni, Cintia, Sedano Tous, María J, Querol, Luis, Illa, Isabel, Wang, Yuzhong, Nobile-Orazio, Eduardo, Rinaldi, Simon, Schenone, Angelo, Pardo, Julio, Vermeij, Frederique H, Lehmann, Helmar C, Granit, Volkan, Cavaletti, Guido, Gutiérrez-Gutiérrez, Gerardo, Barroso, Fabio A, Visser, Leo H, Katzberg, Hans D, Dardiotis, Efthimios, Attarian, Shahram, van der Kooi, Anneke J, Eftimov, Filip, Wirtz, Paul W, Samijn, Johnny P A, Gilhuis, H Jacobus, Hadden, Robert D M, Holt, James K L, Sheikh, Kazim A, Karafiath, Summer, Vytopil, Michal, Antonini, Giovanni, Feasby, Thomas E, Faber, Catharina G, Gijsbers, Cees J, Busby, Mark, Roberts, Rhys C, Silvestri, Nicholas J, Fazio, Raffaella, van Dijk, Gert W, Garssen, Marcel P J, Straathof, Chiara S M, Gorson, Kenneth C, and Jacobs, Bart C

Abstract

Background and objectives: The clinical course and outcome of the Guillain-Barré syndrome (GBS) are diverse and vary among regions. The modified Erasmus GBS Outcome Score (mEGOS), developed with data from Dutch patients, is a clinical model that predicts the risk of walking inability in patients with GBS. The study objective was to validate the mEGOS in the International GBS Outcome Study (IGOS) cohort and to improve its performance and region specificity. Methods: We used prospective data from the first 1,500 patients included in IGOS, aged ≥6 years and unable to walk independently. We evaluated whether the mEGOS at entry and week 1 could predict the inability to walk unaided at 4 and 26 weeks in the full cohort and in regional subgroups, using 2 measures for model performance: (1) discrimination: area under the receiver operating characteristic curve (AUC) and (2) calibration: observed vs predicted probability of being unable to walk independently. To improve the model predictions, we recalibrated the model containing the overall mEGOS score, without changing the individual predictive factors. Finally, we assessed the predictive ability of the individual factors. Results: For validation of mEGOS at entry, 809 patients were eligible (Europe/North America [n = 677], Asia [n = 76], other [n = 56]), and 671 for validation of mEGOS at week 1 (Europe/North America [n = 563], Asia [n = 65], other [n = 43]). AUC values were >0.7 in all regional subgroups. In the Europe/North America subgroup, observed outcomes were worse than predicted; in Asia, observed outcomes were better than predicted. Recalibration improved model accuracy and enabled the development of a region-specific version for Europe/North America (mEGOS-Eu/NA). Similar to the original mEGOS, severe limb weakness and higher age were the predominant predictors of poor outcome in the IGOS cohort. Discussion: mEGOS is a validated tool to predict the inability to walk unaided at 4 and 26 weeks in patients with

Published
2022

59. Functional disorders as a common motor manifestation of COVID‐19 infection or vaccination.

Author

Fung, Wilson K. W., Sa'di, Qais, Katzberg, Hans, Chen, Robert, Lang, Anthony E., Cheung, Angela M., and Fasano, Alfonso

Subjects
SARS-CoV-2, COVID-19, COVID-19 vaccines, MOVEMENT disorders, MENTAL illness, NEUROLOGIC manifestations of general diseases
Abstract

Background and purpose: There have been over 500 million confirmed cases of severe acute respiratory syndrome coronavirus‐2 (SARS‐CoV‐2), also known as coronavirus disease 2019 (COVID‐19), across the globe. To date, a broad spectrum of neurological manifestations following acute infections as well as COVID‐19 vaccines have been reported. The aim of this study was to describe the spectrum of neurological manifestations seen in the 'COVID‐19 clinic' established in a tertiary Movement Disorders clinic. Methods: In this consecutive case‐series study over the period March 2020–January 2022, clinical information regarding demographic data, clinical history and examination findings, investigation results and video recordings of outpatients with motor manifestations associated with COVID‐19 infection or vaccination were reviewed. Results: Twenty‐one adult patients were reviewed in this ad hoc clinic at Toronto Western Hospital. The majority of the patients were female (76%) and the mean (range) age was 50.7 ± 17.2 (21–80) years. Nine patients (43%) presented with motor manifestations following COVID‐19 infection. Twelve patients (57%) developed neurological symptoms following at least one dose of the mRNA or viral vector‐based COVID‐19 vaccine. The most common manifestation observed was a functional movement disorder (43%). The vaccine group demonstrated a higher number of functional disorders compared to the infection group (58% vs. 22%; p = 0.08). Conclusion: Functional motor manifestations can be associated with COVID‐19 and are likely to be under‐reported. In view of the co‐existence of functional symptoms, movement disorders and mental health conditions observed in this study, we would advocate the use of dedicated COVID‐19 Neurology clinics with full access to an experienced multidisciplinary team. [ABSTRACT FROM AUTHOR]

Published
2023
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66. Comparing the NIS vs. MRC and INCAT sensory scale through Rasch analyses

Author

Draak, Thomas H. P., Vanhoutte, Els K., van Nes, Sonja I., Gorson, Kenneth C., Van der Pol, W.-Ludo, Notermans, Nicolette C., Nobile-Orazio, Eduardo, Lewis, Richard A., Léger, Jean-Marc, Van den Bergh, Peter Y. K., Lauria, Giuseppe, Bril, Vera, Katzberg, Hans, Lunn, Michael P. T., Pouget, Jean, van der Kooi, Anneke J., Hahn, Angelika F., van den Berg, Leonard H., van Doorn, Pieter A., Cornblath, David R., Faber, Catharina G., Merkies, Ingemar S. J., Barreira, A. A., Bennett, D., Hadden, R. D., Hughes, R. A.C., Lunn, M. P.T., Reilly, M. M., van den Berg, L. H., van Doorn, P. A., Faber, C. G., van der Kooi, A. J., Merkies, I. S.J., Notermans, N. C., Raaphorts, J., van Schaik, I. N., de Visser, M., Cats, E. A., Van den Bergh, P. Y.K., Bombelli, F., Costa, R., Franques, J., Léger, J-M., Pouget, J., Bril, V., Hahn, A. F., Katzberg, H., Campanella, A., Devigili, G., Gallia, F., Lauria, G., Nobile-Orazio, E., Padua, L., Cornblath, D. R., Gorson, K. C., Lewis, R. A., Illa, I, Querol, L., and van Nes, S. I.

Published
2015
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67. Grip strength comparison in immune-mediated neuropathies: Vigorimeter vs. Jamar

Author

Draak, Thomas H. P., Pruppers, Mariëlle H. J., van Nes, Sonja I., Vanhoutte, Els K., Bakkers, Mayienne, Gorson, Kenneth C., Van der Pol, W.-Ludo, Lewis, Richard. A., Notermans, Nicolette C., Nobile-Orazio, Eduardo, Léger, Jean-Marc, Van den Bergh, Peter Y. K., Lauria, Giuseppe, Bril, Vera, Katzberg, Hans, Lunn, Michael P. T., Pouget, Jean, van der Kooi, Anneke J., van den Berg, Leonard H., van Doorn, Pieter A., Cornblath, David R., Hahn, Angelika F., Faber, Catharina G., Merkies, Ingemar S. J., Barreira, A. A., Bennett, D., Hadden, R. D., Hughes, R. A.C., Lunn, M. P.T., Reilly, M. M., van den Berg, L. H., van Doorn, P. A., Faber, C. G., van der Kooi, A. J., Merkies, I. S.J., Notermans, N. C., Raaphorts, J., van Schaik, I. N., de Visser, M., Cats, E. A., Van den Bergh, P. Y.K., Bombelli, F., Costa, R., Franques, J., Léger, J-M., Pouget, J., Bril, V., Hahn, A. F., Katzberg, H., Campanella, A., Devigili, G., Gallia, F., Lauria, G., Nobile-Orazio, E., Padua, L., Cornblath, D. R., Gorson, K. C., Lewis, R. A., Illa, I, Querol, L., and van Nes, S. I.

Published
2015
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68. Impairment measures versus inflammatory RODS in GBS and CIDP: a responsiveness comparison

Author

Vanhoutte, Els K., Draak, Thomas H. P., Gorson, Kenneth C., van Nes, Sonja I., Hoeijmakers, Janneke G. J., Van der Pol, W.-Ludo, Notermans, Nicolette. C., Lewis, Richard A., Nobile-Orazio, Eduardo, Léger, Jean-Marc, Van den Bergh, Peter Y. K., Lauria, Giuseppe, Bril, Vera, Katzberg, Hans, Lunn, Michael P. T., Pouget, Jean, van der Kooi, Anneke J., Hahn, Angelika F., van Doorn, Pieter A., Cornblath, David R., van den Berg, Leonard H., Faber, Catharina G., Merkies, Ingemar S. J., Barreira, A. A., Bennett, D., Hadden, R. D., Hughes, R. A.C., Lunn, M. P.T., Reilly, M. M., van den Berg, L. H., van Doorn, P. A., Faber, C. G., van der Kooi, A. J., Merkies, I. S.J., Notermans, N. C., Raaphorts, J., van Schaik, I. N., de Visser, M., Cats, E. A., Van den Bergh, P. Y.K., Bombelli, F., Costa, R., Franques, J., Léger, J-M., Pouget, J., Bril, V., Hahn, A. F., Katzberg, H., Campanella, A., Devigili, G., Gallia, F., Lauria, G., Nobile-Orazio, E., Padua, L., Cornblath, D. R., Gorson, K. C., Lewis, R. A., Illa, I, Querol, L., and van Nes, S. I.

Published
2015
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72. Safety, efficacy, and tolerability of efgartigimod in patients with generalised myasthenia gravis (ADAPT): a multicentre, randomised, placebo-controlled, phase 3 trial

Author

Howard, James F, primary, Bril, Vera, additional, Vu, Tuan, additional, Karam, Chafic, additional, Peric, Stojan, additional, Margania, Temur, additional, Murai, Hiroyuki, additional, Bilinska, Malgorzata, additional, Shakarishvili, Roman, additional, Smilowski, Marek, additional, Guglietta, Antonio, additional, Ulrichts, Peter, additional, Vangeneugden, Tony, additional, Utsugisawa, Kimiaki, additional, Verschuuren, Jan, additional, Mantegazza, Renato, additional, De Bleecker, Jan L., additional, De Koning, Kathy, additional, De Mey, Katrien, additional, De Pue, Annelien, additional, Mercelis, Rudolf, additional, Wyckmans, Maren, additional, Vinck, Caroline, additional, Wagemaekers, Linda, additional, Baets, Jonathan, additional, Ng, Eduardo, additional, Shabanpour, Jafar, additional, Daniyal, Lubna, additional, Mannan, Shabber, additional, Katzberg, Hans D., additional, Genge, Angela, additional, Siddiqi, Zaeem, additional, Junkerová, Jana, additional, Horakova, Jana, additional, Reguliova, Katerina, additional, Tyblova, Michaela, additional, Jurajdova, Ivana, additional, Novakova, Iveta, additional, Jakubikova, Michala, additional, Pitha, Jiri, additional, Vohanka, Stanislav, additional, Havelkova, Katerina, additional, Horak, Tomas, additional, Bednarik, Josef, additional, Horakova, Mageda, additional, Meisel, Andreas, additional, Remstedt, Dike, additional, Heibutzki, Claudia, additional, Kohler, Siegfried, additional, Gerischer, Lea, additional, Hoffman, Sarah, additional, Stascheit, Frauke, additional, Vissing, John, additional, Zafirakos, Lizzie, additional, Khatri, Kuldeep Kumar, additional, Autzen, Anne, additional, Godtfeldt Stemmerik, Mads Peter, additional, Andersen, Henning, additional, Attarian, Shahram, additional, Salort-Campana, Emmanuelle, additional, Delmont, Emilien, additional, Grapperon, Aude-Marie, additional, Kouton, Ludivine, additional, Tsiskaridze, Alexander, additional, Rózsa, Csilla, additional, Jakab, Gedeonne Margo, additional, Toth, Szilvia, additional, Szabo, Gyorgyi, additional, Bors, David, additional, Szabo, Eniko, additional, Campanella, Angela, additional, Vanoli, Fiammetta, additional, Frangiamore, Rita, additional, Antozzi, Carlo, additional, Bonanno, Silvia, additional, Maggi, Lorenzo, additional, Giossi, Riccardo, additional, Saccà, Francesco, additional, Marsili, Angela, additional, Pane, Chiara, additional, Puorro, Giorgia, additional, Reia, Antonio, additional, Antonini, Giovanni, additional, Alfieri, Girolamo, additional, Morino, Stefania, additional, Garibaldi, Matteo, additional, Fionda, Laura, additional, Leonardi, Luca, additional, Konno, Shingo, additional, Uzawa, Akiyuki, additional, Sakuma, Kaoru, additional, Watanabe, Chiho, additional, Ozawa, Yukiko, additional, Yasuda, Manato, additional, Onishi, Yosuke, additional, Samukawa, Makoto, additional, Tsuda, Tomoko, additional, Suzuki, Yasushi, additional, Ishida, Sayaka, additional, Watanabe, Genya, additional, Takahashi, Masanori, additional, Nakamura, Hiroko, additional, Sugano, Erina, additional, Kubota, Tomoya, additional, Imai, Tomihiro, additional, Suzuki., Mari, additional, Mori, Ayako, additional, Yamamoto, Daisuke, additional, Ikeda, Kazuna, additional, Hisahara, Shin, additional, Masuda, Masayuki, additional, Takaki, Miki, additional, Minemoto, Kanako, additional, Ido, Nobuhiro, additional, Naito, Makiko, additional, Okubo, Yoshihiko, additional, Sugimoto, Takamichi, additional, Takematsu, Yuka, additional, Kamei, Ayumi, additional, Shimizu, Mihiro, additional, Naito, Hiroyuki, additional, Nomura, Eiichi, additional, Van Heur, Marjolein, additional, Peters, Anne-Marie, additional, Tannemaat, Martijn, additional, Ruiter, Annabel, additional, Keene, Kevin, additional, Halas, Marek, additional, Szczudlik, Andrzej, additional, Pinkosz, Marta, additional, Frasinska, Monika, additional, Zwolinska, Grazyna, additional, Kostera-Pruszczyk, Anna, additional, Golenia, Aleksandra, additional, Szczudlik, Piotr, additional, Szczechowski, Lech, additional, Pasko, Aneta, additional, Poverennova, Irina, additional, Urtaeva, Lubov, additional, Kuznetsova, Nadezhda, additional, Romanova, Tatiana, additional, Nadezhda, Malkova, additional, Lapochka, Elena, additional, Korobko, Denis, additional, Vergunova, Ilona, additional, Melnikova, Anna, additional, Bulatova, Ekaterina, additional, Antipenko, Elena, additional, Basta, Ivana, additional, Bozovic, Ivo, additional, Lavrnic, Dragana, additional, Stojanovic, Vidosava Rakocevic, additional, Beydoun, Said, additional, Akhter, Salma, additional, Malekniazi, Ali, additional, Darki, Leila, additional, Pimentel, Norianne, additional, Cannon, Victoria, additional, Chopra, Manisha, additional, Traub, Rebecca, additional, Mozaffar, Tahseen, additional, Hernandez, Isela, additional, Turner, Ivonne, additional, Habib, Ali, additional, Goyal, Namita, additional, Kak, Manisha, additional, Velasquez, Erik, additional, Lam, Lucy, additional, Suresh, Niraja, additional, Farias, Jerrica, additional, Jones, Sarah, additional, Wagoner, Mary, additional, Eggleston, Debbie, additional, Bertorini, Tulio, additional, Benzel, Cindy, additional, Henegar, Robert, additional, Pillai, Rekha, additional, Bharavaju-Sanka, Ratna, additional, Paiz, Carolyn, additional, Jackson, Carlayne, additional, Ruzhansky, Katherine, additional, Dimitrova, Diana, additional, Visser, Amy, additional, Chahin, Nizar, additional, Levine, Todd, additional, Lisak, Robert, additional, Jia, Kelly, additional, Mada, Flicia, additional, Bernitsas, Evanthia, additional, Pasnoor, Mamatha, additional, Roath, Katherine, additional, Colgan, Samantha, additional, Currence, Melissa, additional, Heim, Andrew, additional, Barohn, Richard, additional, Dimachkie, Mazen, additional, Statland, Jeffrey, additional, Jawdat, Omar, additional, Jabari, Duaa, additional, Farmakidis, Constantine, additional, Gilchrist, James, additional, Li, Yuebing, additional, Caristo, Irys, additional, Hastings, Debbie, additional, Morren, John Anthony, additional, Weiss, Michael, additional, Muppidi, Srikanth, additional, Nguyen, Tia, additional, Welsh, Lesly, additional, So, Yuen, additional, Goyal, Neelam, additional, Pulley, Michael, additional, Bailey, Cathy, additional, Quraishi, Zubair, additional, Berger, Alan, additional, Sahagian, Gregory, additional, Camberos, Yasmin, additional, and Frishberg, Benjamin, additional

Published
2021
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74. Characteristics and Outcomes of Patients with Cerebral Venous Sinus Thrombosis in SARS-CoV-2 Vaccine-Induced Immune Thrombotic Thrombocytopenia

Author

Sánchez Van Kammen, Mayte, Aguiar De Sousa, Diana, Poli, Sven, Cordonnier, Charlotte, Heldner, Mirjam R., Van De Munckhof, Anita, Krzywicka, Katarzyna, Van Haaps, Thijs, Ciccone, Alfonso, Middeldorp, Saskia, Levi, Marcel M., Kremer Hovinga, Johanna A., Silvis, Suzanne, Hiltunen, Sini, Mansour, Maryam, Arauz, Antonio, Barboza, Miguel A., Field, Thalia S., Tsivgoulis, Georgios, Nagel, Simon, Lindgren, Erik, Tatlisumak, Turgut, Jood, Katarina, Putaala, Jukka, Ferro, Jose M., Arnold, Marcel, Coutinho, Jonathan M., Sharma, Aarti R., Elkady, Ahmed, Negro, Alberto, Günther, Albrecht, Gutschalk, Alexander, Schönenberger, Silvia, Buture, Alina, Murphy, Sean, Paiva Nunes, Ana, Tiede, Andreas, Puthuppallil Philip, Anemon, Mengel, Annerose, Medina, Antonio, Hellström Vogel, Åslög, Tawa, Audrey, Aujayeb, Avinash, Casolla, Barbara, Buck, Brian, Zanferrari, Carla, Garcia-Esperon, Carlos, Vayne, Caroline, Legault, Catherine, Pfrepper, Christian, Tracol, Clement, Soriano, Cristina, Guisado-Alonso, Daniel, Bougon, David, Zimatore, Domenico S., Michalski, Dominik, Blacquiere, Dylan, Johansson, Elias, Cuadrado-Godia, Elisa, De Maistre, Emmanuel, Carrera, Emmanuel, Vuillier, Fabrice, Bonneville, Fabrice, Giammello, Fabrizio, Bode, Felix J., Zimmerman, Julian, D'Onofrio, Florindo, Grillo, Francesco, Cotton, Francois, Caparros, François, Puy, Laurent, Maier, Frank, Gulli, Giosue, Frisullo, Giovanni, Polkinghorne, Gregory, Franchineau, Guillaume, Cangür, Hakan, Katzberg, Hans, Sibon, Igor, Baharoglu, Irem, Brar, Jaskiran, Payen, Jean-François, Burrow, Jim, Fernandes, João, Schouten, Judith, Althaus, Katharina, Garambois, Katia, Derex, Laurent, Humbertjean, Lisa, Lebrato Hernandez, Lucia, Kellermair, Lukas, Morin Martin, Mar, Petruzzellis, Marco, Cotelli, Maria, Dubois, Marie-Cécile, Carvalho, Marta, Wittstock, Matthias, Miranda, Miguel, Skjelland, Mona, Bandettini Di Poggio, Monica, Scholz, Moritz J., Raposo, Nicolas, Kahnis, Robert, Kruyt, Nyika, Huet, Olivier, Sharma, Pankaj, Candelaresi, Paolo, Reiner, Peggy, Vieira, Ricardo, Acampora, Roberto, Kern, Rolf, Leker, Ronen, Coutts, Shelagh, Bal, Simerpreet, Sharma, Shyam S., Susen, Sophie, Cox, Thomas, Geeraerts, Thomas, Gattringer, Thomas, Bartsch, Thorsten, Kleinig, Timothy J., Dizonno, Vanessa, Arslan, Yildiz, Sánchez Van Kammen, Mayte, Aguiar De Sousa, Diana, Poli, Sven, Cordonnier, Charlotte, Heldner, Mirjam R., Van De Munckhof, Anita, Krzywicka, Katarzyna, Van Haaps, Thijs, Ciccone, Alfonso, Middeldorp, Saskia, Levi, Marcel M., Kremer Hovinga, Johanna A., Silvis, Suzanne, Hiltunen, Sini, Mansour, Maryam, Arauz, Antonio, Barboza, Miguel A., Field, Thalia S., Tsivgoulis, Georgios, Nagel, Simon, Lindgren, Erik, Tatlisumak, Turgut, Jood, Katarina, Putaala, Jukka, Ferro, Jose M., Arnold, Marcel, Coutinho, Jonathan M., Sharma, Aarti R., Elkady, Ahmed, Negro, Alberto, Günther, Albrecht, Gutschalk, Alexander, Schönenberger, Silvia, Buture, Alina, Murphy, Sean, Paiva Nunes, Ana, Tiede, Andreas, Puthuppallil Philip, Anemon, Mengel, Annerose, Medina, Antonio, Hellström Vogel, Åslög, Tawa, Audrey, Aujayeb, Avinash, Casolla, Barbara, Buck, Brian, Zanferrari, Carla, Garcia-Esperon, Carlos, Vayne, Caroline, Legault, Catherine, Pfrepper, Christian, Tracol, Clement, Soriano, Cristina, Guisado-Alonso, Daniel, Bougon, David, Zimatore, Domenico S., Michalski, Dominik, Blacquiere, Dylan, Johansson, Elias, Cuadrado-Godia, Elisa, De Maistre, Emmanuel, Carrera, Emmanuel, Vuillier, Fabrice, Bonneville, Fabrice, Giammello, Fabrizio, Bode, Felix J., Zimmerman, Julian, D'Onofrio, Florindo, Grillo, Francesco, Cotton, Francois, Caparros, François, Puy, Laurent, Maier, Frank, Gulli, Giosue, Frisullo, Giovanni, Polkinghorne, Gregory, Franchineau, Guillaume, Cangür, Hakan, Katzberg, Hans, Sibon, Igor, Baharoglu, Irem, Brar, Jaskiran, Payen, Jean-François, Burrow, Jim, Fernandes, João, Schouten, Judith, Althaus, Katharina, Garambois, Katia, Derex, Laurent, Humbertjean, Lisa, Lebrato Hernandez, Lucia, Kellermair, Lukas, Morin Martin, Mar, Petruzzellis, Marco, Cotelli, Maria, Dubois, Marie-Cécile, Carvalho, Marta, Wittstock, Matthias, Miranda, Miguel, Skjelland, Mona, Bandettini Di Poggio, Monica, Scholz, Moritz J., Raposo, Nicolas, Kahnis, Robert, Kruyt, Nyika, Huet, Olivier, Sharma, Pankaj, Candelaresi, Paolo, Reiner, Peggy, Vieira, Ricardo, Acampora, Roberto, Kern, Rolf, Leker, Ronen, Coutts, Shelagh, Bal, Simerpreet, Sharma, Shyam S., Susen, Sophie, Cox, Thomas, Geeraerts, Thomas, Gattringer, Thomas, Bartsch, Thorsten, Kleinig, Timothy J., Dizonno, Vanessa, and Arslan, Yildiz

Abstract

Importance: Thrombosis with thrombocytopenia syndrome (TTS) has been reported after vaccination with the SARS-CoV-2 vaccines ChAdOx1 nCov-19 (Oxford-AstraZeneca) and Ad26.COV2.S (Janssen/Johnson & Johnson). Objective: To describe the clinical characteristics and outcome of patients with cerebral venous sinus thrombosis (CVST) after SARS-CoV-2 vaccination with and without TTS. Design, Setting, and Participants: This cohort study used data from an international registry of consecutive patients with CVST within 28 days of SARS-CoV-2 vaccination included between March 29 and June 18, 2021, from 81 hospitals in 19 countries. For reference, data from patients with CVST between 2015 and 2018 were derived from an existing international registry. Clinical characteristics and mortality rate were described for adults with (1) CVST in the setting of SARS-CoV-2 vaccine-induced immune thrombotic thrombocytopenia, (2) CVST after SARS-CoV-2 vaccination not fulling criteria for TTS, and (3) CVST unrelated to SARS-CoV-2 vaccination. Exposures: Patients were classified as having TTS if they had new-onset thrombocytopenia without recent exposure to heparin, in accordance with the Brighton Collaboration interim criteria. Main Outcomes and Measures: Clinical characteristics and mortality rate. Results: Of 116 patients with postvaccination CVST, 78 (67.2%) had TTS, of whom 76 had been vaccinated with ChAdOx1 nCov-19; 38 (32.8%) had no indication of TTS. The control group included 207 patients with CVST before the COVID-19 pandemic. A total of 63 of 78 (81%), 30 of 38 (79%), and 145 of 207 (70.0%) patients, respectively, were female, and the mean (SD) age was 45 (14), 55 (20), and 42 (16) years, respectively. Concomitant thromboembolism occurred in 25 of 70 patients (36%) in the TTS group, 2 of 35 (6%) in the no TTS group, and 10 of 206 (4.9%) in the control group, and in-hospital mortality rates were 47% (36 of 76; 95% CI, 37-58), 5% (2 of 37; 95% CI, 1-18), and 3.9% (8 of 207; 95% CI

Published
2021
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82. Canadian Guidelines for Hereditary Transthyretin Amyloidosis Polyneuropathy Management

Author

Alcantara, Monica, primary, Mezei, Michelle M., additional, Baker, Steven K., additional, Breiner, Ari, additional, Dhawan, Priya, additional, Fiander, Amanda, additional, Fine, Nowell M., additional, Hahn, Christopher, additional, Katzberg, Hans D., additional, Khayambashi, Shahin, additional, Massie, Rami, additional, Matte, Genevieve, additional, Putko, Brendan, additional, Siddiqi, Zaeem, additional, Delgado, Diego, additional, and Bril, Vera, additional

Published
2021
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90. An International Perspective on Preceding Infections in Guillain-Barré Syndrome

Author

Leonhard, Sonja E., van der Eijk, Annemiek A., Andersen, Henning, Antonini, Giovanni, Arends, Samuel, Attarian, Shahram, Barroso, Fabio A., Bateman, Kathleen J., Batstra, Manou R., Benedetti, Luana, van den Berg, Bianca, Van den Bergh, Peter, Bürmann, Jan, Busby, Mark, Casasnovas, Carlos, Cornblath, David R., Davidson, Amy, Doets, Alex Y., van Doorn, Pieter A., Dornonville de la Cour, Charlotte, Feasby, Thomas E., Fehmi, Janev, Garcia-Sobrino, Tania, Goldstein, Jonathan M., Gorson, Kenneth C., Granit, Volkan, Hadden, Robert D.M., Harbo, Thomas, Hartung, Hans-Peter, Hasan, Imran, Holbech, Jakob V., Holt, James K.L., Jahan, Israt, Islam, Zhahirul, Karafiath, Summer, Katzberg, Hans D., Kleyweg, Ruud P., Kolb, Noah, Kuitwaard, Krista, Kuwahara, Motoi, Kusunoki, Susumu, Luijten, Linda W.G., Kuwabara, Satoshi, Lee Pan, Edward, Lehmann, Helmar C., Maas, Marijke, Martín-Aguilar, Lorena, Miller, James A.L., Mohammad, Quazi Deen, Monges, Soledad, Nedkova-Hristova, Velina, Nobile-Orazio, Eduardo, Pardo, Julio, Pereon, Yann, Querol, Luis, Reisin, Ricardo, Van Rijs, Wouter, Rinaldi, Simon, Roberts, Rhys C., Roodbol, Joyce, Shahrizaila, Nortina, Sindrup, Søren Hein, Stein, Beth, Cheng-Yin, Tan, Tankisi, Hatice, Tio-Gillen, Anne P., Sedano Tous, María J., Verboon, Christine, Vermeij, Frederique H., Visser, Leo H., Huizinga, Ruth, Willison, Hugh J., and Jacobs, Bart C.

Published
2022
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92. Reply

Author

Katzberg, Hans D., Barnett, Carolina, and Bril, Vera

Published
2012
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93. Randomized trial of three IVIg doses for treating chronic inflammatory demyelinating polyneuropathy.

Author

Cornblath, David R, Doorn, Pieter A van, Hartung, Hans-Peter, Merkies, Ingemar S J, Katzberg, Hans D, Hinterberger, Doris, Clodi, Elisabeth, Investigators, the ProCID, van Doorn, Pieter A, and ProCID Investigators

Subjects
RESEARCH, CLINICAL trials, RESEARCH methodology, EVALUATION research, TREATMENT effectiveness, COMPARATIVE studies, RANDOMIZED controlled trials, GUILLAIN-Barre syndrome, BLIND experiment, LONGITUDINAL method
Abstract

Intravenous immunoglobulin treatment for chronic inflammatory demyelinating polyneuropathy usually starts with a 2.0 g/kg induction dose followed by 1.0 g/kg maintenance doses every 3 weeks. No dose-ranging studies with intravenous immunoglobulin maintenance therapy have been published. The Progress in Chronic Inflammatory Demyelinating polyneuropathy (ProCID) study was a prospective, double-blind, randomized, parallel-group, multicentre, phase III study investigating the efficacy and safety of 10% liquid intravenous immunoglobulin (Panzyga®) in patients with active chronic inflammatory demyelinating polyneuropathy. Patients were randomized 1:2:1 to receive the standard intravenous immunoglobulin induction dose and then either 0.5, 1.0 or 2.0 g/kg maintenance doses every 3 weeks. The primary end point was the response rate in the 1.0 g/kg group, defined as an improvement ≥1 point in adjusted Inflammatory Neuropathy Cause and Treatment score at Week 6 versus baseline and maintained at Week 24. Secondary end points included dose response and safety. This trial was registered with EudraCT (Number 2015-005443-14) and clinicaltrials.gov (NCT02638207). Between August 2017 and September 2019, the study enrolled 142 patients. All 142 were included in the safety analyses. As no post-infusion data were available for three patients, 139 were included in the efficacy analyses, of whom 121 were previously on corticosteroids. The response rate was 80% (55/69 patients) [95% confidence interval (CI): 69-88%] in the 1.0 g/kg group, 65% (22/34; CI: 48-79%) in the 0.5 g/kg group, and 92% (33/36; CI: 78-97%) in the 2.0 g/kg group. While the proportion of responders was higher with higher maintenance doses, logistic regression analysis showed that the effect on response rate was driven by a significant difference between the 0.5 and 2.0 g/kg groups, whereas the response rates in the 0.5 and 2.0 g/kg groups did not differ significantly from the 1.0 g/kg group. Fifty-six per cent of all patients had an adjusted Inflammatory Neuropathy Cause and Treatment score improvement 3 weeks after the induction dose alone. Treatment-related adverse events were reported in 16 (45.7%), 32 (46.4%) and 20 (52.6%) patients in the 0.5, 1.0 and 2.0 g/kg dose groups, respectively. The most common adverse reaction was headache. There were no treatment-related deaths. Intravenous immunoglobulin (1.0 g/kg) was efficacious and well tolerated as maintenance treatment for patients with chronic inflammatory demyelinating polyneuropathy. Further studies of different maintenance doses of intravenous immunoglobulin in chronic inflammatory demyelinating polyneuropathy are warranted. [ABSTRACT FROM AUTHOR]

Published
2022
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