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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...
Autores principales: | , , |
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Formato: | Online Artículo Texto |
Lenguaje: | English |
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S. Karger AG
2021
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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. |
format | Online Article Text |
id | pubmed-8077499 |
institution | National Center for Biotechnology Information |
language | English |
publishDate | 2021 |
publisher | S. Karger AG |
record_format | MEDLINE/PubMed |
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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