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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...
Autores principales: | , , , |
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Formato: | Online Artículo Texto |
Lenguaje: | English |
Publicado: |
Cold Spring Harbor Laboratory Press
2018
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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. |
format | Online Article Text |
id | pubmed-6169829 |
institution | National Center for Biotechnology Information |
language | English |
publishDate | 2018 |
publisher | Cold Spring Harbor Laboratory Press |
record_format | MEDLINE/PubMed |
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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