Refractory anti-NMDAR encephalitis successfully treated with bortezomib and associated movements disorders controlled with tramadol: a case report with literature review

被引:22
作者
Lazzarin, Serena Marita [1 ,2 ]
Vabanesi, Marco [1 ,2 ]
Cecchetti, Giordano [1 ,2 ]
Fazio, Raffaella [1 ]
Fanelli, Giovanna Franca [2 ]
Volonte, Maria Antonietta [1 ]
Genchi, Angela [1 ]
Giordano, Antonino [1 ]
Martinelli, Vittorio [1 ]
Colombo, Sergio [3 ]
Beccaria, Paolo [3 ]
Mucci, Milena [3 ]
Peccatori, Jacopo [4 ]
Filippi, Massimo [1 ,2 ,5 ,6 ]
Minicucci, Fabio [2 ]
机构
[1] IRCCS, Neurol Unit, San Raffaele Sci Inst, Milan, Italy
[2] IRCCS, Neurophysiol Unit, San Raffaele Sci Inst, Milan, Italy
[3] IRCCS, Gen Intens Care Unit, San Raffaele Sci Inst, Milan, Italy
[4] IRCCS, Hematol & Bone Marrow Transplantat Unit, San Raffaele Sci Inst, Milan, Italy
[5] IRCCS, Inst Expt Neurol, Div Neurosci, Neuroimaging Res Unit,San Raffaele Sci Inst, Via Olgettina 60, I-20132 Milan, Italy
[6] Univ Vita Salute San Raffaele, Milan, Italy
关键词
Anti-NMDAR encephalitis; Autoimmune encephalitis; Bortezomib; EEG; Movement disorders; DEPLETES PLASMA-CELLS; RECEPTOR ENCEPHALITIS; MULTIPLE-MYELOMA; EFFICACY; DYSKINESIA; MANAGEMENT; SPECTRUM; THERAPY; SAFETY;
D O I
10.1007/s00415-020-09988-w
中图分类号
R74 [神经病学与精神病学];
学科分类号
摘要
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.
引用
收藏
页码:2462 / 2468
页数:7
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