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A novel 11p13 microdeletion encompassing PAX6 in a Chinese Han family with aniridia, ptosis and mental retardation

PURPOSE: To explore possible genetic aberrations in a Chinese family with aniridia, ptosis and mental retardation, and provide genetic evidence for the prenatal diagnosis. METHODS: 14 exons of PAX6 in the proband were sequenced by the Sanger sequencing technique. Multiplex ligation-dependent probe a...

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Autores principales: Hu, Ping, Meng, Lulu, Ma, Dingyuan, Qiao, Fengchang, Wang, Yan, Zhou, Jing, Yi, Long, Xu, Zhengfeng
Formato: Online Artículo Texto
Lenguaje:English
Publicado: BioMed Central 2015
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC4307215/
https://www.ncbi.nlm.nih.gov/pubmed/25628759
http://dx.doi.org/10.1186/s13039-015-0110-2
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author Hu, Ping
Meng, Lulu
Ma, Dingyuan
Qiao, Fengchang
Wang, Yan
Zhou, Jing
Yi, Long
Xu, Zhengfeng
author_facet Hu, Ping
Meng, Lulu
Ma, Dingyuan
Qiao, Fengchang
Wang, Yan
Zhou, Jing
Yi, Long
Xu, Zhengfeng
author_sort Hu, Ping
collection PubMed
description PURPOSE: To explore possible genetic aberrations in a Chinese family with aniridia, ptosis and mental retardation, and provide genetic evidence for the prenatal diagnosis. METHODS: 14 exons of PAX6 in the proband were sequenced by the Sanger sequencing technique. Multiplex ligation-dependent probe amplification (MLPA) technique was employed to further explore gene alterations of PAX6. Single nucleotide polymorphisms-array (SNP-array) assay was applied to screen potential pathologic genome-wide copy number variations (CNV). RESULTS: There were no detectable pathogenic mutations in the 14 exons of PAX6 in the proband. MLPA indicated a heterozygous deletion encompassing all PAX6 gene regions covered and a partial upstream region. SNP-array assay detected a heterozygous 11p13 microdeletion with a length of 518 kb in the proband, spanning two whole annotated genes, elongation factor protein 4 (ELP4), the paired box gene 6 (PAX6), and partial IMP1 inner-mitochondrial membrane (IMMP1L) gene. SNP-array revealed her affected brother carried the identical deletion. CONCLUSIONS: The 518 kb heterozygous deletion in 11p13 encompassing PAX6 should be the genetic etiology for the familial aniridia.
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spelling pubmed-43072152015-01-28 A novel 11p13 microdeletion encompassing PAX6 in a Chinese Han family with aniridia, ptosis and mental retardation Hu, Ping Meng, Lulu Ma, Dingyuan Qiao, Fengchang Wang, Yan Zhou, Jing Yi, Long Xu, Zhengfeng Mol Cytogenet Case Report PURPOSE: To explore possible genetic aberrations in a Chinese family with aniridia, ptosis and mental retardation, and provide genetic evidence for the prenatal diagnosis. METHODS: 14 exons of PAX6 in the proband were sequenced by the Sanger sequencing technique. Multiplex ligation-dependent probe amplification (MLPA) technique was employed to further explore gene alterations of PAX6. Single nucleotide polymorphisms-array (SNP-array) assay was applied to screen potential pathologic genome-wide copy number variations (CNV). RESULTS: There were no detectable pathogenic mutations in the 14 exons of PAX6 in the proband. MLPA indicated a heterozygous deletion encompassing all PAX6 gene regions covered and a partial upstream region. SNP-array assay detected a heterozygous 11p13 microdeletion with a length of 518 kb in the proband, spanning two whole annotated genes, elongation factor protein 4 (ELP4), the paired box gene 6 (PAX6), and partial IMP1 inner-mitochondrial membrane (IMMP1L) gene. SNP-array revealed her affected brother carried the identical deletion. CONCLUSIONS: The 518 kb heterozygous deletion in 11p13 encompassing PAX6 should be the genetic etiology for the familial aniridia. BioMed Central 2015-01-22 /pmc/articles/PMC4307215/ /pubmed/25628759 http://dx.doi.org/10.1186/s13039-015-0110-2 Text en © Hu et al.; licensee BioMed Central. 2015 This is an Open Access article distributed under the terms of the Creative Commons Attribution License (http://creativecommons.org/licenses/by/4.0), which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly credited. The Creative Commons Public Domain Dedication waiver (http://creativecommons.org/publicdomain/zero/1.0/) applies to the data made available in this article, unless otherwise stated.
spellingShingle Case Report
Hu, Ping
Meng, Lulu
Ma, Dingyuan
Qiao, Fengchang
Wang, Yan
Zhou, Jing
Yi, Long
Xu, Zhengfeng
A novel 11p13 microdeletion encompassing PAX6 in a Chinese Han family with aniridia, ptosis and mental retardation
title A novel 11p13 microdeletion encompassing PAX6 in a Chinese Han family with aniridia, ptosis and mental retardation
title_full A novel 11p13 microdeletion encompassing PAX6 in a Chinese Han family with aniridia, ptosis and mental retardation
title_fullStr A novel 11p13 microdeletion encompassing PAX6 in a Chinese Han family with aniridia, ptosis and mental retardation
title_full_unstemmed A novel 11p13 microdeletion encompassing PAX6 in a Chinese Han family with aniridia, ptosis and mental retardation
title_short A novel 11p13 microdeletion encompassing PAX6 in a Chinese Han family with aniridia, ptosis and mental retardation
title_sort novel 11p13 microdeletion encompassing pax6 in a chinese han family with aniridia, ptosis and mental retardation
topic Case Report
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC4307215/
https://www.ncbi.nlm.nih.gov/pubmed/25628759
http://dx.doi.org/10.1186/s13039-015-0110-2
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