Bortezomib-Responsive Refractory Anti-N-Methyl-D-Aspartate Receptor Encephalitis

被引：25

作者：

Cordani, Ramona ^{[1
]}

Micalizzi, Concetta ^{[2
]}

Giacomini, Thea ^{[1
]}

Gastaldi, Matteo ^{[3
]}

Franciotta, Diego ^{[3
]}

Fioredda, Francesca ^{[2
]}

Buratti, Silvia ^{[4
]}

Morana, Giovanni ^{[5
]}

Pirlo, Daniela ^{[6
]}

Renna, Salvatore ^{[6
]}

Castagnola, Elio ^{[7
]}

Risso, Marco ^{[8
]}

Lanteri, Paola ^{[9
]}

Vari, Maria Stella ^{[10
]}

Mancardi, Maria Margherita ^{[11
]}

机构：

[1] Univ Genoa, IRCCS Giannina Gaslini, Dept Neurosci Ophthalmol Genet & Maternal Infanti, Unit Child Neuropsichiat, Genoa, Italy

[2] IRCCS Giannina Gaslini, Unit Hematol, Genoa, Italy

[3] IRCCS Mondino Fdn, Neuroimmunol Lab, Pavia, Italy

[4] IRCCS Giannina Gaslini, Neonatal & Pediat Intens Care Unit, Genoa, Italy

[5] IRCCS Giannina Gaslini, Unit Neuroradiol, Genoa, Italy

[6] IRCCS Giannina Gaslini, Dept Emergency, Genoa, Italy

[7] IRCCS Giannina Gaslini, Infect Dis Unit, Genoa, Italy

[8] IRCCS Giannina Gaslini, Unit Immunol & Transfus Med, Genoa, Italy

[9] IRCCS Giannina Gaslini, Unit Neurophysiol, Genoa, Italy

[10] IRCCS Giannina Gaslini, Unit Pediat Neurol & Neurogenerat Dis, Genoa, Italy

[11] IRCCS Giannina Gaslini, Dept Clin & Surg Neurosci & Rehabil, Unit Child Neuropsichiat, Genoa, Italy

来源：

PEDIATRIC NEUROLOGY | 2020年 / 103卷

关键词：

Autoimmune encephalitis; Anti-N-methyl-D-aspartate receptor (NMDAR) antibodies; Immunosuppressive therapy; Proteasome inhibitor; Cell-based assay; Arterial spin labeling (ASL); Refractory status epilepticus;

D O I：

10.1016/j.pediatrneurol.2019.09.004

中图分类号：

R74 [神经病学与精神病学];

学科分类号：

摘要：

Background: Anti-N-methyl-D-aspartate receptor encephalitis is a central nervous system inflammatory autoimmune disease affecting adults and children. The use of first- and second-line immunotherapies is supported. Recent reports suggest the efficacy of bortezomib in severe anti-N-methyl-D-aspartate encephalitis in adult patients not responsive to second-line treatment; there are no data about pediatric patients. patient description: We describe an eight-year-old child with anti-N-methyl-D-aspartate encephalitis not responsive to first- and second-line treatments who experienced marked clinical improvement after bortezomib administration. Discussion: Bortezomib is a selective and reversible inhibitor of the 26S proteasome, which is used to treat oncologic and rare autoimmune disorders in pediatric patients. As observed in adult patients, bortezomib administration induced anti-N-methyl-D-aspartate antibody titer decline and clinical improvement with an acceptable risk profile. Conclusion: This is the first report of the use of bortezomib in children with anti-N-methyl-D-aspartate encephalitis; it could be a useful therapeutic option in children with refractory anti-N-methyl-D-aspartate encephalitis. (C) 2019 Elsevier Inc. All rights reserved.

引用

页码：61 / 64

页数：4

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