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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...

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Autores principales: Traylor, Ryan N, Fan, Zheng, Hudson, Beth, Rosenfeld, Jill A, Shaffer, Lisa G, Torchia, Beth S, Ballif, Blake C
Formato: Texto
Lenguaje:English
Publicado: BioMed Central 2009
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.
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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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