Document Detail


HOXA1 mutations are not a common cause of Möbius syndrome.
MedLine Citation:
PMID:  20227628     Owner:  NLM     Status:  MEDLINE    
Abstract/OtherAbstract:
The HOXA1-related syndromes result from autosomal-recessive truncating mutations in the homeobox transcription factor, HOXA1. Limited horizontal gaze and sensorineural deafness are the most common features; affected individuals can also have facial weakness, mental retardation, autism, motor disabilities, central hypoventilation, carotid artery, and/or conotruncal heart defects. Möbius syndrome is also phenotypically heterogeneous, with minimal diagnostic criteria of nonprogressive facial weakness and impaired ocular abduction; mental retardation, autism, motor disabilities, additional eye movements restrictions, hearing loss, hypoventilation, and craniofacial, lingual, and limb abnormalities also occur. We asked, given the phenotypic overlap between these syndromes and the variable expressivity of both disorders, whether individuals with Möbius syndrome might harbor mutations in HOXA1. Our results suggest that HOXA1 mutations are not a common cause of sporadic Möbius syndrome in the general population.
Authors:
Jessica K Rankin; Caroline Andrews; Wai-Man Chan; Elizabeth C Engle
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Publication Detail:
Type:  Journal Article; Research Support, N.I.H., Extramural; Research Support, Non-U.S. Gov't    
Journal Detail:
Title:  Journal of AAPOS : the official publication of the American Association for Pediatric Ophthalmology and Strabismus / American Association for Pediatric Ophthalmology and Strabismus     Volume:  14     ISSN:  1528-3933     ISO Abbreviation:  J AAPOS     Publication Date:  2010 Feb 
Date Detail:
Created Date:  2010-03-15     Completed Date:  2010-06-11     Revised Date:  2010-12-03    
Medline Journal Info:
Nlm Unique ID:  9710011     Medline TA:  J AAPOS     Country:  United States    
Other Details:
Languages:  eng     Pagination:  78-80     Citation Subset:  IM    
Copyright Information:
Copyright (c) 2010 American Association for Pediatric Ophthalmology and Strabismus. Published by Mosby, Inc. All rights reserved.
Affiliation:
Department of Neurology, Children's Hospital Boston, Boston, Massachusetts, USA.
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MeSH Terms
Descriptor/Qualifier:
Child
Duane Retraction Syndrome / genetics*
Exons / genetics
Genetic Predisposition to Disease
Homeodomain Proteins / genetics*
Humans
Introns / genetics
Mobius Syndrome / genetics*
Phenotype
Polymorphism, Single Nucleotide*
Transcription Factors / genetics*
Grant Support
ID/Acronym/Agency:
HD18655/HD/NICHD NIH HHS; NEI R01EY15298/EY/NEI NIH HHS; P30 HD018655-28/HD/NICHD NIH HHS; R01 EY015298-05/EY/NEI NIH HHS; //Howard Hughes Medical Institute
Chemical
Reg. No./Substance:
0/Homeodomain Proteins; 0/Transcription Factors; 0/homeobox A1 protein
Comments/Corrections

From MEDLINE®/PubMed®, a database of the U.S. National Library of Medicine


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