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Microdeletion of 6q16.1 encompassing EPHA7 in a child with mild neurological abnormalities and dysmorphic features: case report
BACKGROUND: Of the fewer than 100 cases reported within the literature of constitutional deletions involving the long arm of chromosome 6, only five have been characterized using high-resolution microarray analysis. Reported 6q deletion patients show a high incidence of mental retardation, ear anoma...
Autores principales: | , , , , , , |
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Formato: | Texto |
Lenguaje: | English |
Publicado: |
BioMed Central
2009
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Materias: | |
Acceso en línea: | https://www.ncbi.nlm.nih.gov/pmc/articles/PMC2731778/ https://www.ncbi.nlm.nih.gov/pubmed/19664229 http://dx.doi.org/10.1186/1755-8166-2-17 |
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author | Traylor, Ryan N Fan, Zheng Hudson, Beth Rosenfeld, Jill A Shaffer, Lisa G Torchia, Beth S Ballif, Blake C |
author_facet | Traylor, Ryan N Fan, Zheng Hudson, Beth Rosenfeld, Jill A Shaffer, Lisa G Torchia, Beth S Ballif, Blake C |
author_sort | Traylor, Ryan N |
collection | PubMed |
description | BACKGROUND: Of the fewer than 100 cases reported within the literature of constitutional deletions involving the long arm of chromosome 6, only five have been characterized using high-resolution microarray analysis. Reported 6q deletion patients show a high incidence of mental retardation, ear anomalies, hypotonia, and postnatal growth retardation. RESULTS: We report a 16-month-old male presenting with developmental delay and dysmorphic features who was found by array-based comparative genomic hybridization (aCGH) to have a ~2.16 Mb de novo deletion within chromosome band 6q16.1 that encompasses only two genes. Expression studies of the mouse homologue of one of the genes, the ephrin receptor 7 gene (EPHA7), have shown the gene functions during murine embryogenesis to form cortical domains, determine brain size and shape, and play a role in development of the central nervous system (CNS). DISCUSSION: Our results suggest that deletion of EPHA7 plays a role in the neurologic and dysmorphic features, including developmental delay, hypotonia, and ear malformations, observed in some 6q deletion patients. |
format | Text |
id | pubmed-2731778 |
institution | National Center for Biotechnology Information |
language | English |
publishDate | 2009 |
publisher | BioMed Central |
record_format | MEDLINE/PubMed |
spelling | pubmed-27317782009-08-26 Microdeletion of 6q16.1 encompassing EPHA7 in a child with mild neurological abnormalities and dysmorphic features: case report Traylor, Ryan N Fan, Zheng Hudson, Beth Rosenfeld, Jill A Shaffer, Lisa G Torchia, Beth S Ballif, Blake C Mol Cytogenet Case Report BACKGROUND: Of the fewer than 100 cases reported within the literature of constitutional deletions involving the long arm of chromosome 6, only five have been characterized using high-resolution microarray analysis. Reported 6q deletion patients show a high incidence of mental retardation, ear anomalies, hypotonia, and postnatal growth retardation. RESULTS: We report a 16-month-old male presenting with developmental delay and dysmorphic features who was found by array-based comparative genomic hybridization (aCGH) to have a ~2.16 Mb de novo deletion within chromosome band 6q16.1 that encompasses only two genes. Expression studies of the mouse homologue of one of the genes, the ephrin receptor 7 gene (EPHA7), have shown the gene functions during murine embryogenesis to form cortical domains, determine brain size and shape, and play a role in development of the central nervous system (CNS). DISCUSSION: Our results suggest that deletion of EPHA7 plays a role in the neurologic and dysmorphic features, including developmental delay, hypotonia, and ear malformations, observed in some 6q deletion patients. BioMed Central 2009-08-07 /pmc/articles/PMC2731778/ /pubmed/19664229 http://dx.doi.org/10.1186/1755-8166-2-17 Text en Copyright © 2009 Traylor et al; licensee BioMed Central Ltd. http://creativecommons.org/licenses/by/2.0 This is an Open Access article distributed under the terms of the Creative Commons Attribution License ( (http://creativecommons.org/licenses/by/2.0) ), which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited. |
spellingShingle | Case Report Traylor, Ryan N Fan, Zheng Hudson, Beth Rosenfeld, Jill A Shaffer, Lisa G Torchia, Beth S Ballif, Blake C Microdeletion of 6q16.1 encompassing EPHA7 in a child with mild neurological abnormalities and dysmorphic features: case report |
title | Microdeletion of 6q16.1 encompassing EPHA7 in a child with mild neurological abnormalities and dysmorphic features: case report |
title_full | Microdeletion of 6q16.1 encompassing EPHA7 in a child with mild neurological abnormalities and dysmorphic features: case report |
title_fullStr | Microdeletion of 6q16.1 encompassing EPHA7 in a child with mild neurological abnormalities and dysmorphic features: case report |
title_full_unstemmed | Microdeletion of 6q16.1 encompassing EPHA7 in a child with mild neurological abnormalities and dysmorphic features: case report |
title_short | Microdeletion of 6q16.1 encompassing EPHA7 in a child with mild neurological abnormalities and dysmorphic features: case report |
title_sort | microdeletion of 6q16.1 encompassing epha7 in a child with mild neurological abnormalities and dysmorphic features: case report |
topic | Case Report |
url | https://www.ncbi.nlm.nih.gov/pmc/articles/PMC2731778/ https://www.ncbi.nlm.nih.gov/pubmed/19664229 http://dx.doi.org/10.1186/1755-8166-2-17 |
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