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Biallelic variants in VARS in a family with two siblings with intellectual disability and microcephaly: case report and review of the literature

Two male siblings ages 15 and 10 yr old had similar features of intellectual disability, developmental delay, severe speech impairment, microcephaly, prematurity, and transient elevation of liver enzymes in infancy. Exome sequencing revealed one novel (c.65C>A; p.Ala22Asp) and one ultra-rare (c.3...

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Detalles Bibliográficos
Autores principales: Okur, Volkan, Ganapathi, Mythily, Wilson, Ashley, Chung, Wendy K.
Formato: Online Artículo Texto
Lenguaje:English
Publicado: Cold Spring Harbor Laboratory Press 2018
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC6169829/
https://www.ncbi.nlm.nih.gov/pubmed/30275004
http://dx.doi.org/10.1101/mcs.a003301
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author Okur, Volkan
Ganapathi, Mythily
Wilson, Ashley
Chung, Wendy K.
author_facet Okur, Volkan
Ganapathi, Mythily
Wilson, Ashley
Chung, Wendy K.
author_sort Okur, Volkan
collection PubMed
description Two male siblings ages 15 and 10 yr old had similar features of intellectual disability, developmental delay, severe speech impairment, microcephaly, prematurity, and transient elevation of liver enzymes in infancy. Exome sequencing revealed one novel (c.65C>A; p.Ala22Asp) and one ultra-rare (c.3214T>C; p.Phe1072Leu) predicted damaging missense variant in trans in the gene encoding cytoplasmic valyl-tRNA synthetase (VARS). Biallelic variants in VARS have previously been associated with a neurodevelopmental disorder characterized by microcephaly, seizures, and cortical atrophy (NDMSCA; MIM #617802). Although our patients have no history of seizures or cortical atrophy, we suggest that the biallelic variants in VARS p.Ala22Asp and p.Phe1072Leu in this family are likely pathogenic and associated with NDMSCA, expanding the clinical phenotype of the condition.
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spelling pubmed-61698292018-10-12 Biallelic variants in VARS in a family with two siblings with intellectual disability and microcephaly: case report and review of the literature Okur, Volkan Ganapathi, Mythily Wilson, Ashley Chung, Wendy K. Cold Spring Harb Mol Case Stud Rapid Communication Two male siblings ages 15 and 10 yr old had similar features of intellectual disability, developmental delay, severe speech impairment, microcephaly, prematurity, and transient elevation of liver enzymes in infancy. Exome sequencing revealed one novel (c.65C>A; p.Ala22Asp) and one ultra-rare (c.3214T>C; p.Phe1072Leu) predicted damaging missense variant in trans in the gene encoding cytoplasmic valyl-tRNA synthetase (VARS). Biallelic variants in VARS have previously been associated with a neurodevelopmental disorder characterized by microcephaly, seizures, and cortical atrophy (NDMSCA; MIM #617802). Although our patients have no history of seizures or cortical atrophy, we suggest that the biallelic variants in VARS p.Ala22Asp and p.Phe1072Leu in this family are likely pathogenic and associated with NDMSCA, expanding the clinical phenotype of the condition. Cold Spring Harbor Laboratory Press 2018-10 /pmc/articles/PMC6169829/ /pubmed/30275004 http://dx.doi.org/10.1101/mcs.a003301 Text en © 2018 Okur et al.; Published by Cold Spring Harbor Laboratory Press http://creativecommons.org/licenses/by-nc/4.0/ This article is distributed under the terms of the Creative Commons Attribution-NonCommercial License (http://creativecommons.org/licenses/by-nc/4.0/) , which permits reuse and redistribution, except for commercial purposes, provided that the original author and source are credited.
spellingShingle Rapid Communication
Okur, Volkan
Ganapathi, Mythily
Wilson, Ashley
Chung, Wendy K.
Biallelic variants in VARS in a family with two siblings with intellectual disability and microcephaly: case report and review of the literature
title Biallelic variants in VARS in a family with two siblings with intellectual disability and microcephaly: case report and review of the literature
title_full Biallelic variants in VARS in a family with two siblings with intellectual disability and microcephaly: case report and review of the literature
title_fullStr Biallelic variants in VARS in a family with two siblings with intellectual disability and microcephaly: case report and review of the literature
title_full_unstemmed Biallelic variants in VARS in a family with two siblings with intellectual disability and microcephaly: case report and review of the literature
title_short Biallelic variants in VARS in a family with two siblings with intellectual disability and microcephaly: case report and review of the literature
title_sort biallelic variants in vars in a family with two siblings with intellectual disability and microcephaly: case report and review of the literature
topic Rapid Communication
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC6169829/
https://www.ncbi.nlm.nih.gov/pubmed/30275004
http://dx.doi.org/10.1101/mcs.a003301
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