Cargando…

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...

Descripción completa

Detalles Bibliográficos
Autores principales: Dell'Isola, Giovanni Battista, Vinti, Valerio, Fattorusso, Antonella, Tascini, Giorgia, Mencaroni, Elisabetta, Di Cara, Giuseppe, Striano, Pasquale, Verrotti, Alberto
Formato: Online Artículo Texto
Lenguaje:English
Publicado: Frontiers Media S.A. 2022
Materias:
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
_version_ 1784642491857764352
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
work_keys_str_mv AT dellisolagiovannibattista thebroadclinicalspectrumofepilepsiesassociatedwithprotocadherin19genemutation
AT vintivalerio thebroadclinicalspectrumofepilepsiesassociatedwithprotocadherin19genemutation
AT fattorussoantonella thebroadclinicalspectrumofepilepsiesassociatedwithprotocadherin19genemutation
AT tascinigiorgia thebroadclinicalspectrumofepilepsiesassociatedwithprotocadherin19genemutation
AT mencaronielisabetta thebroadclinicalspectrumofepilepsiesassociatedwithprotocadherin19genemutation
AT dicaragiuseppe thebroadclinicalspectrumofepilepsiesassociatedwithprotocadherin19genemutation
AT strianopasquale thebroadclinicalspectrumofepilepsiesassociatedwithprotocadherin19genemutation
AT verrottialberto thebroadclinicalspectrumofepilepsiesassociatedwithprotocadherin19genemutation
AT dellisolagiovannibattista broadclinicalspectrumofepilepsiesassociatedwithprotocadherin19genemutation
AT vintivalerio broadclinicalspectrumofepilepsiesassociatedwithprotocadherin19genemutation
AT fattorussoantonella broadclinicalspectrumofepilepsiesassociatedwithprotocadherin19genemutation
AT tascinigiorgia broadclinicalspectrumofepilepsiesassociatedwithprotocadherin19genemutation
AT mencaronielisabetta broadclinicalspectrumofepilepsiesassociatedwithprotocadherin19genemutation
AT dicaragiuseppe broadclinicalspectrumofepilepsiesassociatedwithprotocadherin19genemutation
AT strianopasquale broadclinicalspectrumofepilepsiesassociatedwithprotocadherin19genemutation
AT verrottialberto broadclinicalspectrumofepilepsiesassociatedwithprotocadherin19genemutation