PMID- 32535682 OWN - NLM STAT- MEDLINE DCOM- 20210618 LR - 20210618 IS - 1432-1459 (Electronic) IS - 0340-5354 (Linking) VI - 267 IP - 8 DP - 2020 Aug TI - Refractory anti-NMDAR encephalitis successfully treated with bortezomib and associated movements disorders controlled with tramadol: a case report with literature review. PG - 2462-2468 LID - 10.1007/s00415-020-09988-w [doi] AB - 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. FAU - Lazzarin, Serena Marita AU - Lazzarin SM AD - Neurology Unit, IRCCS San Raffaele Scientific Institute, Milan, Italy. AD - Neurophysiology Unit, IRCCS San Raffaele Scientific Institute, Milan, Italy. FAU - Vabanesi, Marco AU - Vabanesi M AUID- ORCID: 0000-0002-8283-7079 AD - Neurology Unit, IRCCS San Raffaele Scientific Institute, Milan, Italy. AD - Neurophysiology Unit, IRCCS San Raffaele Scientific Institute, Milan, Italy. FAU - Cecchetti, Giordano AU - Cecchetti G AUID- ORCID: 0000-0002-6544-410X AD - Neurology Unit, IRCCS San Raffaele Scientific Institute, Milan, Italy. AD - Neurophysiology Unit, IRCCS San Raffaele Scientific Institute, Milan, Italy. FAU - Fazio, Raffaella AU - Fazio R AD - Neurology Unit, IRCCS San Raffaele Scientific Institute, Milan, Italy. FAU - Fanelli, Giovanna Franca AU - Fanelli GF AD - Neurophysiology Unit, IRCCS San Raffaele Scientific Institute, Milan, Italy. FAU - Volonte, Maria Antonietta AU - Volonte MA AD - Neurology Unit, IRCCS San Raffaele Scientific Institute, Milan, Italy. FAU - Genchi, Angela AU - Genchi A AD - Neurology Unit, IRCCS San Raffaele Scientific Institute, Milan, Italy. FAU - Giordano, Antonino AU - Giordano A AUID- ORCID: 0000-0002-3685-8311 AD - Neurology Unit, IRCCS San Raffaele Scientific Institute, Milan, Italy. FAU - Martinelli, Vittorio AU - Martinelli V AUID- ORCID: 0000-0002-5987-5739 AD - Neurology Unit, IRCCS San Raffaele Scientific Institute, Milan, Italy. FAU - Colombo, Sergio AU - Colombo S AD - General Intensive Care Unit, IRCCS San Raffaele Scientific Institute, Milan, Italy. FAU - Beccaria, Paolo AU - Beccaria P AD - General Intensive Care Unit, IRCCS San Raffaele Scientific Institute, Milan, Italy. FAU - Mucci, Milena AU - Mucci M AD - General Intensive Care Unit, IRCCS San Raffaele Scientific Institute, Milan, Italy. FAU - Peccatori, Jacopo AU - Peccatori J AD - Hematology and Bone Marrow Transplantation Unit, IRCCS San Raffaele Scientific Institute, Milan, Italy. FAU - Filippi, Massimo AU - Filippi M AUID- ORCID: 0000-0002-5485-0479 AD - Neurology Unit, IRCCS San Raffaele Scientific Institute, Milan, Italy. filippi.massimo@hsr.it. AD - Neurophysiology Unit, IRCCS San Raffaele Scientific Institute, Milan, Italy. filippi.massimo@hsr.it. AD - 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. AD - Vita-Salute San Raffaele University, Milan, Italy. filippi.massimo@hsr.it. FAU - Minicucci, Fabio AU - Minicucci F AD - Neurophysiology Unit, IRCCS San Raffaele Scientific Institute, Milan, Italy. LA - eng PT - Case Reports PT - Journal Article PT - Review DEP - 20200613 PL - Germany TA - J Neurol JT - Journal of neurology JID - 0423161 RN - 39J1LGJ30J (Tramadol) RN - 4F4X42SYQ6 (Rituximab) RN - 69G8BD63PP (Bortezomib) SB - IM EIN - J Neurol. 2021 Feb;268(2):741-742. PMID: 33417007 MH - Adult MH - *Anti-N-Methyl-D-Aspartate Receptor Encephalitis/complications/drug therapy MH - Bortezomib/therapeutic use MH - Female MH - Humans MH - *Movement Disorders/drug therapy/etiology MH - Rituximab MH - *Tramadol OTO - NOTNLM OT - Anti-NMDAR encephalitis OT - Autoimmune encephalitis OT - Bortezomib OT - EEG OT - Movement disorders EDAT- 2020/06/15 06:00 MHDA- 2021/06/22 06:00 CRDT- 2020/06/15 06:00 PHST- 2020/04/16 00:00 [received] PHST- 2020/06/09 00:00 [accepted] PHST- 2020/06/08 00:00 [revised] PHST- 2020/06/15 06:00 [pubmed] PHST- 2021/06/22 06:00 [medline] PHST- 2020/06/15 06:00 [entrez] AID - 10.1007/s00415-020-09988-w [pii] AID - 10.1007/s00415-020-09988-w [doi] PST - ppublish SO - J Neurol. 2020 Aug;267(8):2462-2468. doi: 10.1007/s00415-020-09988-w. Epub 2020 Jun 13.