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The Broad Clinical Spectrum of Epilepsies Associated With Protocadherin 19 Gene Mutation
Protocadherin 19 (PCDH19) gene is one of the most common genes involved in epilepsy syndromes. According to literature data PCDH19 is among the 6 genes most involved in genetic epilepsies. PCDH19 is located on chromosome Xq22.1 and is involved in neuronal connections and signal transduction. The mos...
Autores principales: | , , , , , , , |
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Formato: | Online Artículo Texto |
Lenguaje: | English |
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Frontiers Media S.A.
2022
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Acceso en línea: | https://www.ncbi.nlm.nih.gov/pmc/articles/PMC8801579/ https://www.ncbi.nlm.nih.gov/pubmed/35111125 http://dx.doi.org/10.3389/fneur.2021.780053 |
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author | Dell'Isola, Giovanni Battista Vinti, Valerio Fattorusso, Antonella Tascini, Giorgia Mencaroni, Elisabetta Di Cara, Giuseppe Striano, Pasquale Verrotti, Alberto |
author_facet | Dell'Isola, Giovanni Battista Vinti, Valerio Fattorusso, Antonella Tascini, Giorgia Mencaroni, Elisabetta Di Cara, Giuseppe Striano, Pasquale Verrotti, Alberto |
author_sort | Dell'Isola, Giovanni Battista |
collection | PubMed |
description | Protocadherin 19 (PCDH19) gene is one of the most common genes involved in epilepsy syndromes. According to literature data PCDH19 is among the 6 genes most involved in genetic epilepsies. PCDH19 is located on chromosome Xq22.1 and is involved in neuronal connections and signal transduction. The most frequent clinical expression of PCDH19 mutation is epilepsy and mental retardation limited to female (EFMR) characterized by epileptic and non-epileptic symptoms affecting mainly females. However, the phenotypic spectrum of these mutations is considerably variable from genetic epilepsy with febrile seizure plus to epileptic encephalopathies. The peculiar exclusive involvement of females seems to be caused by a cellular interference in heterozygosity, however, affected mosaic-males have been reported. Seizure types range from focal seizure to generalized tonic-clonic, tonic, atonic, absences, and myoclonic jerks. Treatment of PCDH19-related epilepsy is limited by drug resistance and by the absence of specific treatment indications. However, seizures become less severe with adolescence and some patients may even become seizure-free. Non-epileptic symptoms represent the main disabilities of adult patients with PCDH19 mutation. This review aims to analyze the highly variable phenotypic expression of PCDH19 gene mutation associated with epilepsy. |
format | Online Article Text |
id | pubmed-8801579 |
institution | National Center for Biotechnology Information |
language | English |
publishDate | 2022 |
publisher | Frontiers Media S.A. |
record_format | MEDLINE/PubMed |
spelling | pubmed-88015792022-02-01 The Broad Clinical Spectrum of Epilepsies Associated With Protocadherin 19 Gene Mutation Dell'Isola, Giovanni Battista Vinti, Valerio Fattorusso, Antonella Tascini, Giorgia Mencaroni, Elisabetta Di Cara, Giuseppe Striano, Pasquale Verrotti, Alberto Front Neurol Neurology Protocadherin 19 (PCDH19) gene is one of the most common genes involved in epilepsy syndromes. According to literature data PCDH19 is among the 6 genes most involved in genetic epilepsies. PCDH19 is located on chromosome Xq22.1 and is involved in neuronal connections and signal transduction. The most frequent clinical expression of PCDH19 mutation is epilepsy and mental retardation limited to female (EFMR) characterized by epileptic and non-epileptic symptoms affecting mainly females. However, the phenotypic spectrum of these mutations is considerably variable from genetic epilepsy with febrile seizure plus to epileptic encephalopathies. The peculiar exclusive involvement of females seems to be caused by a cellular interference in heterozygosity, however, affected mosaic-males have been reported. Seizure types range from focal seizure to generalized tonic-clonic, tonic, atonic, absences, and myoclonic jerks. Treatment of PCDH19-related epilepsy is limited by drug resistance and by the absence of specific treatment indications. However, seizures become less severe with adolescence and some patients may even become seizure-free. Non-epileptic symptoms represent the main disabilities of adult patients with PCDH19 mutation. This review aims to analyze the highly variable phenotypic expression of PCDH19 gene mutation associated with epilepsy. Frontiers Media S.A. 2022-01-17 /pmc/articles/PMC8801579/ /pubmed/35111125 http://dx.doi.org/10.3389/fneur.2021.780053 Text en Copyright © 2022 Dell'Isola, Vinti, Fattorusso, Tascini, Mencaroni, Di Cara, Striano and Verrotti. https://creativecommons.org/licenses/by/4.0/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(s) 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. |
spellingShingle | Neurology Dell'Isola, Giovanni Battista Vinti, Valerio Fattorusso, Antonella Tascini, Giorgia Mencaroni, Elisabetta Di Cara, Giuseppe Striano, Pasquale Verrotti, Alberto The Broad Clinical Spectrum of Epilepsies Associated With Protocadherin 19 Gene Mutation |
title | The Broad Clinical Spectrum of Epilepsies Associated With Protocadherin 19 Gene Mutation |
title_full | The Broad Clinical Spectrum of Epilepsies Associated With Protocadherin 19 Gene Mutation |
title_fullStr | The Broad Clinical Spectrum of Epilepsies Associated With Protocadherin 19 Gene Mutation |
title_full_unstemmed | The Broad Clinical Spectrum of Epilepsies Associated With Protocadherin 19 Gene Mutation |
title_short | The Broad Clinical Spectrum of Epilepsies Associated With Protocadherin 19 Gene Mutation |
title_sort | broad clinical spectrum of epilepsies associated with protocadherin 19 gene mutation |
topic | Neurology |
url | https://www.ncbi.nlm.nih.gov/pmc/articles/PMC8801579/ https://www.ncbi.nlm.nih.gov/pubmed/35111125 http://dx.doi.org/10.3389/fneur.2021.780053 |
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