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Mutation in SNAP25 as a novel genetic cause of epilepsy and intellectual disability

Whole exome sequencing using a parent-child trio design to identify de novo mutations provides an efficient method to identify novel genes for rare diseases with low reproductive fitness that are difficult to study by more classical genetic methods of linkage analysis. We describe a 15 y old female...

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Autores principales: Rohena, Luis, Neidich, Julie, Truitt Cho, Megan, Gonzalez, Kelly DF, Tang, Sha, Devinsky, Orrin, Chung, Wendy K
Formato: Online Artículo Texto
Lenguaje:English
Publicado: Landes Bioscience 2013
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC3932847/
https://www.ncbi.nlm.nih.gov/pubmed/25003006
http://dx.doi.org/10.4161/rdis.26314
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author Rohena, Luis
Neidich, Julie
Truitt Cho, Megan
Gonzalez, Kelly DF
Tang, Sha
Devinsky, Orrin
Chung, Wendy K
author_facet Rohena, Luis
Neidich, Julie
Truitt Cho, Megan
Gonzalez, Kelly DF
Tang, Sha
Devinsky, Orrin
Chung, Wendy K
author_sort Rohena, Luis
collection PubMed
description Whole exome sequencing using a parent-child trio design to identify de novo mutations provides an efficient method to identify novel genes for rare diseases with low reproductive fitness that are difficult to study by more classical genetic methods of linkage analysis. We describe a 15 y old female with severe static encephalopathy, intellectual disability, and generalized epilepsy. After extensive metabolic and genetic testing, whole exome sequencing identified a novel de novo variant in Synaptosomal-associated protein-25 (SNAP25), c.142G > T p.Phe48Val alteration. This variant is predicted to be damaging by all prediction algorithms. SNAP25 is part of the soluble N-ethylmaleimide-sensitive factor attachment protein receptor (SNARE) protein complex which is involved in exocytotic release of neurotransmitters. Genetic alterations in Snap25 in animal models can cause anxiety-related behavior, ataxia and seizures. We suggest that SNAP25 mutations in humans are a novel genetic cause of intellectual disability and epilepsy.
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spelling pubmed-39328472014-07-07 Mutation in SNAP25 as a novel genetic cause of epilepsy and intellectual disability Rohena, Luis Neidich, Julie Truitt Cho, Megan Gonzalez, Kelly DF Tang, Sha Devinsky, Orrin Chung, Wendy K Rare Dis Research Paper Whole exome sequencing using a parent-child trio design to identify de novo mutations provides an efficient method to identify novel genes for rare diseases with low reproductive fitness that are difficult to study by more classical genetic methods of linkage analysis. We describe a 15 y old female with severe static encephalopathy, intellectual disability, and generalized epilepsy. After extensive metabolic and genetic testing, whole exome sequencing identified a novel de novo variant in Synaptosomal-associated protein-25 (SNAP25), c.142G > T p.Phe48Val alteration. This variant is predicted to be damaging by all prediction algorithms. SNAP25 is part of the soluble N-ethylmaleimide-sensitive factor attachment protein receptor (SNARE) protein complex which is involved in exocytotic release of neurotransmitters. Genetic alterations in Snap25 in animal models can cause anxiety-related behavior, ataxia and seizures. We suggest that SNAP25 mutations in humans are a novel genetic cause of intellectual disability and epilepsy. Landes Bioscience 2013-09-05 /pmc/articles/PMC3932847/ /pubmed/25003006 http://dx.doi.org/10.4161/rdis.26314 Text en Copyright © 2013 Landes Bioscience http://creativecommons.org/licenses/by-nc/3.0/ This is an open-access article licensed under a Creative Commons Attribution-NonCommercial 3.0 Unported License. The article may be redistributed, reproduced, and reused for non-commercial purposes, provided the original source is properly cited.
spellingShingle Research Paper
Rohena, Luis
Neidich, Julie
Truitt Cho, Megan
Gonzalez, Kelly DF
Tang, Sha
Devinsky, Orrin
Chung, Wendy K
Mutation in SNAP25 as a novel genetic cause of epilepsy and intellectual disability
title Mutation in SNAP25 as a novel genetic cause of epilepsy and intellectual disability
title_full Mutation in SNAP25 as a novel genetic cause of epilepsy and intellectual disability
title_fullStr Mutation in SNAP25 as a novel genetic cause of epilepsy and intellectual disability
title_full_unstemmed Mutation in SNAP25 as a novel genetic cause of epilepsy and intellectual disability
title_short Mutation in SNAP25 as a novel genetic cause of epilepsy and intellectual disability
title_sort mutation in snap25 as a novel genetic cause of epilepsy and intellectual disability
topic Research Paper
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC3932847/
https://www.ncbi.nlm.nih.gov/pubmed/25003006
http://dx.doi.org/10.4161/rdis.26314
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