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Frontal Hypoperfusion and the Effectiveness of Perampanel in Long-Lived Patient with Lafora Disease

We report a long-lived patient with Lafora disease (LD). A 34-year-old woman experienced onset of seizures at the age of 11 years. She was bedridden in her early twenties due to frequent generalized tonic-clonic seizures, myoclonus, and progressive mental deterioration. Her seizures occurred all the...

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Detalles Bibliográficos
Autores principales: Obara, Koji, Abe, Erika, Toyoshima, Itaru
Formato: Online Artículo Texto
Lenguaje:English
Publicado: S. Karger AG 2021
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC8077499/
https://www.ncbi.nlm.nih.gov/pubmed/33976658
http://dx.doi.org/10.1159/000514243
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author Obara, Koji
Abe, Erika
Toyoshima, Itaru
author_facet Obara, Koji
Abe, Erika
Toyoshima, Itaru
author_sort Obara, Koji
collection PubMed
description We report a long-lived patient with Lafora disease (LD). A 34-year-old woman experienced onset of seizures at the age of 11 years. She was bedridden in her early twenties due to frequent generalized tonic-clonic seizures, myoclonus, and progressive mental deterioration. Her seizures occurred all the time despite administration of multiple anticonvulsants at high doses. At the age of 31, she started perampanel, which resulted in reduction of anticonvulsants after her visible myoclonus and convulsions disappeared. Brain magnetic resonance imaging showed marked cerebral and cerebellar atrophy, and single-photon emission computed tomography using N-isopropyl-p-[<sup>123</sup>I] iodoamphetamine (IMP-SPECT) revealed significant hypoperfusion of the frontal lobe and cerebellum. We identified a W219R homozygous mutation in exon 1 of the NHLRC1 gene. Because perampanel may not only control seizures but also prevent mental deterioration in LD, we propose that perampanel should be administered from the early stage of LD.
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spelling pubmed-80774992021-05-10 Frontal Hypoperfusion and the Effectiveness of Perampanel in Long-Lived Patient with Lafora Disease Obara, Koji Abe, Erika Toyoshima, Itaru Case Rep Neurol Single Case − General Neurology We report a long-lived patient with Lafora disease (LD). A 34-year-old woman experienced onset of seizures at the age of 11 years. She was bedridden in her early twenties due to frequent generalized tonic-clonic seizures, myoclonus, and progressive mental deterioration. Her seizures occurred all the time despite administration of multiple anticonvulsants at high doses. At the age of 31, she started perampanel, which resulted in reduction of anticonvulsants after her visible myoclonus and convulsions disappeared. Brain magnetic resonance imaging showed marked cerebral and cerebellar atrophy, and single-photon emission computed tomography using N-isopropyl-p-[<sup>123</sup>I] iodoamphetamine (IMP-SPECT) revealed significant hypoperfusion of the frontal lobe and cerebellum. We identified a W219R homozygous mutation in exon 1 of the NHLRC1 gene. Because perampanel may not only control seizures but also prevent mental deterioration in LD, we propose that perampanel should be administered from the early stage of LD. S. Karger AG 2021-03-22 /pmc/articles/PMC8077499/ /pubmed/33976658 http://dx.doi.org/10.1159/000514243 Text en Copyright © 2021 by S. Karger AG, Basel https://creativecommons.org/licenses/by-nc/4.0/This article is licensed under the Creative Commons Attribution-NonCommercial-4.0 International License (CC BY-NC) (http://www.karger.com/Services/OpenAccessLicense). Usage and distribution for commercial purposes requires written permission.
spellingShingle Single Case − General Neurology
Obara, Koji
Abe, Erika
Toyoshima, Itaru
Frontal Hypoperfusion and the Effectiveness of Perampanel in Long-Lived Patient with Lafora Disease
title Frontal Hypoperfusion and the Effectiveness of Perampanel in Long-Lived Patient with Lafora Disease
title_full Frontal Hypoperfusion and the Effectiveness of Perampanel in Long-Lived Patient with Lafora Disease
title_fullStr Frontal Hypoperfusion and the Effectiveness of Perampanel in Long-Lived Patient with Lafora Disease
title_full_unstemmed Frontal Hypoperfusion and the Effectiveness of Perampanel in Long-Lived Patient with Lafora Disease
title_short Frontal Hypoperfusion and the Effectiveness of Perampanel in Long-Lived Patient with Lafora Disease
title_sort frontal hypoperfusion and the effectiveness of perampanel in long-lived patient with lafora disease
topic Single Case − General Neurology
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC8077499/
https://www.ncbi.nlm.nih.gov/pubmed/33976658
http://dx.doi.org/10.1159/000514243
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