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Refractory anti-NMDAR encephalitis successfully treated with bortezomib and associated movements disorders controlled with tramadol: a case report with literature review.
J Neurol. 2020 Aug; 267(8):2462-2468.JN

Abstract

Anti-N-methyl-D-aspartate receptor (NMDAR) encephalitis is a potentially fatal autoimmune disease, characterized by autoantibody-mediated neurotransmission impairment in multiple brain locations. The course of this condition often comprises altered mental status, autonomic dysfunctions, refractory seizures and hyperkinetic movement disorders. Available disease-modifying therapies include corticosteroids, i.v. immunoglobulins, plasma exchange, rituximab and cyclophosphamide. In a subgroup of patients not responding to B-cell depletion, bortezomib, a proteasome inhibitor, has shown promising evidence of efficacy. The time course of recovery from acute phase may be very slow (weeks/months), and only few data are available in literature about the concurrent management of encephalitis-associated movement disorders. We report a case of severe anti-NMDAR encephalitis in a 29-year-old woman, not responsive to first- and second-line treatments, with persistent involuntary motor manifestations. Starting three months after symptom onset, four cycles of bortezomib have been administered; subsequently we observed a progressive improvement of neurological status. Meanwhile, motor manifestations were controlled after the administration of tramadol, a non-competitive NMDA receptor antagonist.

Authors+Show Affiliations

Neurology Unit, IRCCS San Raffaele Scientific Institute, Milan, Italy. Neurophysiology Unit, IRCCS San Raffaele Scientific Institute, Milan, Italy.Neurology Unit, IRCCS San Raffaele Scientific Institute, Milan, Italy. Neurophysiology Unit, IRCCS San Raffaele Scientific Institute, Milan, Italy.Neurology Unit, IRCCS San Raffaele Scientific Institute, Milan, Italy. Neurophysiology Unit, IRCCS San Raffaele Scientific Institute, Milan, Italy.Neurology Unit, IRCCS San Raffaele Scientific Institute, Milan, Italy.Neurophysiology Unit, IRCCS San Raffaele Scientific Institute, Milan, Italy.Neurology Unit, IRCCS San Raffaele Scientific Institute, Milan, Italy.Neurology Unit, IRCCS San Raffaele Scientific Institute, Milan, Italy.Neurology Unit, IRCCS San Raffaele Scientific Institute, Milan, Italy.Neurology Unit, IRCCS San Raffaele Scientific Institute, Milan, Italy.General Intensive Care Unit, IRCCS San Raffaele Scientific Institute, Milan, Italy.General Intensive Care Unit, IRCCS San Raffaele Scientific Institute, Milan, Italy.General Intensive Care Unit, IRCCS San Raffaele Scientific Institute, Milan, Italy.Hematology and Bone Marrow Transplantation Unit, IRCCS San Raffaele Scientific Institute, Milan, Italy.Neurology Unit, IRCCS San Raffaele Scientific Institute, Milan, Italy. filippi.massimo@hsr.it. Neurophysiology Unit, IRCCS San Raffaele Scientific Institute, Milan, Italy. filippi.massimo@hsr.it. Neuroimaging Research Unit, Institute of Experimental Neurology, Division of Neuroscience, IRCCS San Raffaele Scientific Institute, Via Olgettina, 60, 20132, Milan, Italy. filippi.massimo@hsr.it. Vita-Salute San Raffaele University, Milan, Italy. filippi.massimo@hsr.it.Neurophysiology Unit, IRCCS San Raffaele Scientific Institute, Milan, Italy.

Pub Type(s)

Journal Article

Language

eng

PubMed ID

32535682

Citation

Lazzarin, Serena Marita, et al. "Refractory anti-NMDAR Encephalitis Successfully Treated With Bortezomib and Associated Movements Disorders Controlled With Tramadol: a Case Report With Literature Review." Journal of Neurology, vol. 267, no. 8, 2020, pp. 2462-2468.
Lazzarin SM, Vabanesi M, Cecchetti G, et al. Refractory anti-NMDAR encephalitis successfully treated with bortezomib and associated movements disorders controlled with tramadol: a case report with literature review. J Neurol. 2020;267(8):2462-2468.
Lazzarin, S. M., Vabanesi, M., Cecchetti, G., Fazio, R., Fanelli, G. F., Volonté, M. A., Genchi, A., Giordano, A., Martinelli, V., Colombo, S., Beccaria, P., Mucci, M., Peccatori, J., Filippi, M., & Minicucci, F. (2020). Refractory anti-NMDAR encephalitis successfully treated with bortezomib and associated movements disorders controlled with tramadol: a case report with literature review. Journal of Neurology, 267(8), 2462-2468. https://doi.org/10.1007/s00415-020-09988-w
Lazzarin SM, et al. Refractory anti-NMDAR Encephalitis Successfully Treated With Bortezomib and Associated Movements Disorders Controlled With Tramadol: a Case Report With Literature Review. J Neurol. 2020;267(8):2462-2468. PubMed PMID: 32535682.
* Article titles in AMA citation format should be in sentence-case
TY - JOUR T1 - Refractory anti-NMDAR encephalitis successfully treated with bortezomib and associated movements disorders controlled with tramadol: a case report with literature review. AU - Lazzarin,Serena Marita, AU - Vabanesi,Marco, AU - Cecchetti,Giordano, AU - Fazio,Raffaella, AU - Fanelli,Giovanna Franca, AU - Volonté,Maria Antonietta, AU - Genchi,Angela, AU - Giordano,Antonino, AU - Martinelli,Vittorio, AU - Colombo,Sergio, AU - Beccaria,Paolo, AU - Mucci,Milena, AU - Peccatori,Jacopo, AU - Filippi,Massimo, AU - Minicucci,Fabio, Y1 - 2020/06/13/ PY - 2020/04/16/received PY - 2020/06/09/accepted PY - 2020/06/08/revised PY - 2020/6/15/pubmed PY - 2020/6/15/medline PY - 2020/6/15/entrez KW - Anti-NMDAR encephalitis KW - Autoimmune encephalitis KW - Bortezomib KW - EEG KW - Movement disorders SP - 2462 EP - 2468 JF - Journal of neurology JO - J. Neurol. VL - 267 IS - 8 N2 - Anti-N-methyl-D-aspartate receptor (NMDAR) encephalitis is a potentially fatal autoimmune disease, characterized by autoantibody-mediated neurotransmission impairment in multiple brain locations. The course of this condition often comprises altered mental status, autonomic dysfunctions, refractory seizures and hyperkinetic movement disorders. Available disease-modifying therapies include corticosteroids, i.v. immunoglobulins, plasma exchange, rituximab and cyclophosphamide. In a subgroup of patients not responding to B-cell depletion, bortezomib, a proteasome inhibitor, has shown promising evidence of efficacy. The time course of recovery from acute phase may be very slow (weeks/months), and only few data are available in literature about the concurrent management of encephalitis-associated movement disorders. We report a case of severe anti-NMDAR encephalitis in a 29-year-old woman, not responsive to first- and second-line treatments, with persistent involuntary motor manifestations. Starting three months after symptom onset, four cycles of bortezomib have been administered; subsequently we observed a progressive improvement of neurological status. Meanwhile, motor manifestations were controlled after the administration of tramadol, a non-competitive NMDA receptor antagonist. SN - 1432-1459 UR - https://www.unboundmedicine.com/medline/citation/32535682/Refractory_anti-NMDAR_encephalitis_successfully_treated_with_bortezomib_and_associated_movements_disorders_controlled_with_tramadol:_a_case_report_with_literature_review L2 - https://dx.doi.org/10.1007/s00415-020-09988-w DB - PRIME DP - Unbound Medicine ER -
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