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Unexplained Progressive Neurological Deficits after Corpus Callosotomy May Be Caused by Autoimmune Encephalitis: A Case of Suspected Postoperative Anti-NMDAR Encephalitis

The main causes of anti-N-methyl-D-aspartate receptor (NMDAR) encephalitis are ovarian teratoma and herpes simplex virus (HSV) encephalitis. We present a rare case of suspected anti-NMDAR encephalitis caused by corpus callosotomy (CC). An 18-year-old woman with Lennox-Gastaut syndrome underwent CC....

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Autores principales: Hatano, Keisuke, Fujimoto, Ayataka, Sato, Keishiro, Yamamoto, Takamichi, Sakuma, Hiroshi, Enoki, Hideo
Formato: Online Artículo Texto
Lenguaje:English
Publicado: MDPI 2023
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC9856623/
https://www.ncbi.nlm.nih.gov/pubmed/36672116
http://dx.doi.org/10.3390/brainsci13010135
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author Hatano, Keisuke
Fujimoto, Ayataka
Sato, Keishiro
Yamamoto, Takamichi
Sakuma, Hiroshi
Enoki, Hideo
author_facet Hatano, Keisuke
Fujimoto, Ayataka
Sato, Keishiro
Yamamoto, Takamichi
Sakuma, Hiroshi
Enoki, Hideo
author_sort Hatano, Keisuke
collection PubMed
description The main causes of anti-N-methyl-D-aspartate receptor (NMDAR) encephalitis are ovarian teratoma and herpes simplex virus (HSV) encephalitis. We present a rare case of suspected anti-NMDAR encephalitis caused by corpus callosotomy (CC). An 18-year-old woman with Lennox-Gastaut syndrome underwent CC. Although left hemiplegic due to cerebral hemorrhage and impaired consciousness due to cerebral venous sinus thrombosis (CVST) appeared postoperatively, anticoagulant therapy quickly improved CVST and impaired consciousness. However, various unexplained symptoms such as insomnia, hallucination, impulsivity, impaired consciousness, and a new type of drug-resistant cluster seizures gradually developed over a 2-month period. Magnetic resonance imaging revealed the gradual extension of a hyperintense area from the right frontal lobe on fluid-attenuated inversion recovery images. Intravenous methylprednisolone pulse was initiated from postoperative day (POD) 74, followed by intravenous immunoglobulin (IVIg) therapy, although white blood cell counts were normal in all three cerebrospinal fluid (CSF) examinations. After IVIg therapy, the above unexplained symptoms promptly improved. On POD 103, antibodies against NMDAR were revealed in both the serum and CSF collected before these immunotherapies. The patient was transferred to a rehabilitation hospital due to residual left hemiplegia. Psychiatric symptoms and a new onset of drug-resistant seizures may be suggestive of postoperative anti-NMDAR encephalitis, even if CSF findings are mild.
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spelling pubmed-98566232023-01-21 Unexplained Progressive Neurological Deficits after Corpus Callosotomy May Be Caused by Autoimmune Encephalitis: A Case of Suspected Postoperative Anti-NMDAR Encephalitis Hatano, Keisuke Fujimoto, Ayataka Sato, Keishiro Yamamoto, Takamichi Sakuma, Hiroshi Enoki, Hideo Brain Sci Case Report The main causes of anti-N-methyl-D-aspartate receptor (NMDAR) encephalitis are ovarian teratoma and herpes simplex virus (HSV) encephalitis. We present a rare case of suspected anti-NMDAR encephalitis caused by corpus callosotomy (CC). An 18-year-old woman with Lennox-Gastaut syndrome underwent CC. Although left hemiplegic due to cerebral hemorrhage and impaired consciousness due to cerebral venous sinus thrombosis (CVST) appeared postoperatively, anticoagulant therapy quickly improved CVST and impaired consciousness. However, various unexplained symptoms such as insomnia, hallucination, impulsivity, impaired consciousness, and a new type of drug-resistant cluster seizures gradually developed over a 2-month period. Magnetic resonance imaging revealed the gradual extension of a hyperintense area from the right frontal lobe on fluid-attenuated inversion recovery images. Intravenous methylprednisolone pulse was initiated from postoperative day (POD) 74, followed by intravenous immunoglobulin (IVIg) therapy, although white blood cell counts were normal in all three cerebrospinal fluid (CSF) examinations. After IVIg therapy, the above unexplained symptoms promptly improved. On POD 103, antibodies against NMDAR were revealed in both the serum and CSF collected before these immunotherapies. The patient was transferred to a rehabilitation hospital due to residual left hemiplegia. Psychiatric symptoms and a new onset of drug-resistant seizures may be suggestive of postoperative anti-NMDAR encephalitis, even if CSF findings are mild. MDPI 2023-01-12 /pmc/articles/PMC9856623/ /pubmed/36672116 http://dx.doi.org/10.3390/brainsci13010135 Text en © 2023 by the authors. https://creativecommons.org/licenses/by/4.0/Licensee MDPI, Basel, Switzerland. This article is an open access article distributed under the terms and conditions of the Creative Commons Attribution (CC BY) license (https://creativecommons.org/licenses/by/4.0/).
spellingShingle Case Report
Hatano, Keisuke
Fujimoto, Ayataka
Sato, Keishiro
Yamamoto, Takamichi
Sakuma, Hiroshi
Enoki, Hideo
Unexplained Progressive Neurological Deficits after Corpus Callosotomy May Be Caused by Autoimmune Encephalitis: A Case of Suspected Postoperative Anti-NMDAR Encephalitis
title Unexplained Progressive Neurological Deficits after Corpus Callosotomy May Be Caused by Autoimmune Encephalitis: A Case of Suspected Postoperative Anti-NMDAR Encephalitis
title_full Unexplained Progressive Neurological Deficits after Corpus Callosotomy May Be Caused by Autoimmune Encephalitis: A Case of Suspected Postoperative Anti-NMDAR Encephalitis
title_fullStr Unexplained Progressive Neurological Deficits after Corpus Callosotomy May Be Caused by Autoimmune Encephalitis: A Case of Suspected Postoperative Anti-NMDAR Encephalitis
title_full_unstemmed Unexplained Progressive Neurological Deficits after Corpus Callosotomy May Be Caused by Autoimmune Encephalitis: A Case of Suspected Postoperative Anti-NMDAR Encephalitis
title_short Unexplained Progressive Neurological Deficits after Corpus Callosotomy May Be Caused by Autoimmune Encephalitis: A Case of Suspected Postoperative Anti-NMDAR Encephalitis
title_sort unexplained progressive neurological deficits after corpus callosotomy may be caused by autoimmune encephalitis: a case of suspected postoperative anti-nmdar encephalitis
topic Case Report
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC9856623/
https://www.ncbi.nlm.nih.gov/pubmed/36672116
http://dx.doi.org/10.3390/brainsci13010135
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