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Front. Neurol. | doi: 10.3389/fneur.2018.00081

Management of Refractory Orofacial Dyskinesia Caused by Anti-N-methyl-D-aspartate Receptor Encephalitis using Botulinum Toxin

 Feixia Zheng1*, xiuyun ye1,  xulai shi1,  Neha D. Poonit1 and zhongdong lin1*
  • 1Second Affiliated Hospital of Wenzhou Medical College, China

The use of botulinum toxin type A (BoNT-A) injections for the treatment of orofacial dyskinesia secondary to anti-N-methyl-D-aspartate receptor (NMDAR) encephalitis is rarely reported. Here we report a case of an urgent, successful management of severe orofacial dyskinesia in an 8-year-old girl with anti-NMDAR encephalitis using BoNT-A injection. The patient presented with de novo unilateral paroxysmal movement disorder progressing to generalized dystonia and repetitive orofacial dyskinesia. Diagnosis was confirmed by the presence of NMDAR antibodies in serum and cerebrospinal fluid. The orofacial dyskinesia worsened despite the aggressive use of first-line immunotherapy and second-line immunotherapy (rituximab), and resulted in a potentially fatal self-inflicted oral injury. We urgently attempted symptomatic management using BoNT-A injections in the masseter, and induced muscle paralysis using vecuronium. The patient’s severe orofacial dyskinesia was controlled. We observed the effects of the BoNT-A injections and a tapering off of the effects of vecuronium ten days after the treatment. The movement disorder had improved significantly four weeks after the first administration of rituximab. The injection of BoNT-A into the masseter may be an effective treatment for medically refractory orofacial dyskinesia in pediatric patients with anti-NMDAR encephalitis. We propose that the use of BoNT-A injections should be considered early to avoid self-inflicted oral injury due to severe refractory orofacial dyskinesia in patients with anti-NMDAR encephalitis.

Keywords: Anti-N-Methyl-D-Aspartate Receptor Encephalitis, Dyskinesia, Botulinum Toxin, Stereotypic Movement Disorder, vecuronium, rituximab, Pediatrics

Received: 14 Dec 2017; Accepted: 05 Feb 2018.

Edited by:

Guillaume Sébire, McGill University, Canada

Reviewed by:

Brahim Tabarki, University of Sousse, Tunisia
Domingo Escudero, Hospital Clínic de Barcelona, Spain  

Copyright: © 2018 Zheng, ye, shi, Poonit and lin. This is an open-access article distributed under the terms of the Creative Commons Attribution License (CC BY). The use, distribution or reproduction in other forums is permitted, provided the original author(s) and the copyright owner are credited and that the original publication in this journal is cited, in accordance with accepted academic practice. No use, distribution or reproduction is permitted which does not comply with these terms.

* Correspondence:
Mrs. Feixia Zheng, Second Affiliated Hospital of Wenzhou Medical College, Wenzhou, China, zhfxia06@163.com
Dr. zhongdong lin, Second Affiliated Hospital of Wenzhou Medical College, Wenzhou, China, wzlinzd@163.com