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Pediatric anti-NMDA receptor encephalitis with catatonia: treatment with electroconvulsive therapy.
Pediatr Rheumatol Online J. 2019 Feb 18; 17(1):8.PR

Abstract

BACKGROUND

Anti-NMDA receptor encephalitis, an autoimmune disease associated with antibodies against N-methyl-D-aspartate (NMDA) receptors, is being diagnosed more frequently, especially in children and young adults. Acute neurological and psychiatric manifestations are the common presenting symptoms. Diagnosing anti-NMDA receptor encephalitis is often challenging given the wide range of clinical presentation, and may be further complicated by its overlap of symptoms, brain MRI changes, and CSF findings with other entities affecting the brain. Even though diagnosis can be made by identifying antibodies in immune-mediated encephalitis, the diagnosis may be delayed by weeks to months. Delay in initiation of treatment with immune suppressive therapies is shown to be associated with adverse outcomes. Malignant catatonia is a severe and life-threatening state associated with anti-NMDA receptor encephalitis. It is often inadequately assessed and may not respond to immunosuppressive treatment.

CASE PRESENTATION

We present a confirmed case of anti-NMDA receptor encephalitis in a 16 year old girl who had severe critical neurological and psychiatric manifestations, including malignant catatonia and autonomic instability. Our patient continued to manifest malignant catatonia despite the initiation of prompt, aggressive immune suppressive therapies, including corticosteroids, plasmapheresis, intravenous gammaglobulin and rituximab, as well as treatment with high-dose benzodiazepines. Once electroconvulsive therapy (ECT) began, she had a robust response with resolution of her catatonia. Six weeks after treatment with eight ECT cycles, she had returned to her normal baseline cognitive and motor function.

CONCLUSIONS

ECT was an effective and well-tolerated therapy in our patient, and should be considered for the treatment of children with anti-NMDA receptor encephalitis whose catatonia does not respond to immunosuppression and benzodiazepines.

Authors+Show Affiliations

Section of Pediatric Rheumatology, Department of Pediatrics, University of Chicago Medical Center, 5841 S. Maryland Avenue, MC 5044, Chicago, 60637, IL, USA.Section of Pediatric Psychiatry, Department of Psychiatry, University of Chicago Medical Center, Chicago, USA.Department of Psychiatry, University of Chicago Medical Center, Chicago, USA.Department of Psychiatry, University of Chicago Medical Center, Chicago, USA.Department of Pediatrics, University of Chicago Medical Center, Chicago, USA.Section of Pediatric Rheumatology, Department of Pediatrics, University of Chicago Medical Center, 5841 S. Maryland Avenue, MC 5044, Chicago, 60637, IL, USA. lww@uchicago.edu.

Pub Type(s)

Case Reports
Journal Article

Language

eng

PubMed ID

30777097

Citation

Moussa, Taha, et al. "Pediatric anti-NMDA Receptor Encephalitis With Catatonia: Treatment With Electroconvulsive Therapy." Pediatric Rheumatology Online Journal, vol. 17, no. 1, 2019, p. 8.
Moussa T, Afzal K, Cooper J, et al. Pediatric anti-NMDA receptor encephalitis with catatonia: treatment with electroconvulsive therapy. Pediatr Rheumatol Online J. 2019;17(1):8.
Moussa, T., Afzal, K., Cooper, J., Rosenberger, R., Gerstle, K., & Wagner-Weiner, L. (2019). Pediatric anti-NMDA receptor encephalitis with catatonia: treatment with electroconvulsive therapy. Pediatric Rheumatology Online Journal, 17(1), 8. https://doi.org/10.1186/s12969-019-0310-0
Moussa T, et al. Pediatric anti-NMDA Receptor Encephalitis With Catatonia: Treatment With Electroconvulsive Therapy. Pediatr Rheumatol Online J. 2019 Feb 18;17(1):8. PubMed PMID: 30777097.
* Article titles in AMA citation format should be in sentence-case
TY - JOUR T1 - Pediatric anti-NMDA receptor encephalitis with catatonia: treatment with electroconvulsive therapy. AU - Moussa,Taha, AU - Afzal,Khalid, AU - Cooper,Joseph, AU - Rosenberger,Ryan, AU - Gerstle,Karyn, AU - Wagner-Weiner,Linda, Y1 - 2019/02/18/ PY - 2018/12/11/received PY - 2019/02/07/accepted PY - 2019/2/20/entrez PY - 2019/2/20/pubmed PY - 2019/2/23/medline KW - Anti-NMDA receptor encephalitis KW - Catatonia KW - Corticosteroids KW - Electroconvulsive therapy KW - Intravenous immunoglobulins KW - Plasma exchange KW - Rituximab SP - 8 EP - 8 JF - Pediatric rheumatology online journal JO - Pediatr Rheumatol Online J VL - 17 IS - 1 N2 - BACKGROUND: Anti-NMDA receptor encephalitis, an autoimmune disease associated with antibodies against N-methyl-D-aspartate (NMDA) receptors, is being diagnosed more frequently, especially in children and young adults. Acute neurological and psychiatric manifestations are the common presenting symptoms. Diagnosing anti-NMDA receptor encephalitis is often challenging given the wide range of clinical presentation, and may be further complicated by its overlap of symptoms, brain MRI changes, and CSF findings with other entities affecting the brain. Even though diagnosis can be made by identifying antibodies in immune-mediated encephalitis, the diagnosis may be delayed by weeks to months. Delay in initiation of treatment with immune suppressive therapies is shown to be associated with adverse outcomes. Malignant catatonia is a severe and life-threatening state associated with anti-NMDA receptor encephalitis. It is often inadequately assessed and may not respond to immunosuppressive treatment. CASE PRESENTATION: We present a confirmed case of anti-NMDA receptor encephalitis in a 16 year old girl who had severe critical neurological and psychiatric manifestations, including malignant catatonia and autonomic instability. Our patient continued to manifest malignant catatonia despite the initiation of prompt, aggressive immune suppressive therapies, including corticosteroids, plasmapheresis, intravenous gammaglobulin and rituximab, as well as treatment with high-dose benzodiazepines. Once electroconvulsive therapy (ECT) began, she had a robust response with resolution of her catatonia. Six weeks after treatment with eight ECT cycles, she had returned to her normal baseline cognitive and motor function. CONCLUSIONS: ECT was an effective and well-tolerated therapy in our patient, and should be considered for the treatment of children with anti-NMDA receptor encephalitis whose catatonia does not respond to immunosuppression and benzodiazepines. SN - 1546-0096 UR - https://www.unboundmedicine.com/medline/citation/30777097/Pediatric_anti_NMDA_receptor_encephalitis_with_catatonia:_treatment_with_electroconvulsive_therapy_ L2 - https://ped-rheum.biomedcentral.com/articles/10.1186/s12969-019-0310-0 DB - PRIME DP - Unbound Medicine ER -