Document Detail


Further case of microdeletion of 8q24 with phenotype overlapping Langer-Giedion without TRPS1 deletion.
MedLine Citation:
PMID:  18478595     Owner:  NLM     Status:  MEDLINE    
Abstract/OtherAbstract:
Langer-Giedion syndrome results from a microdeletion at 8q24.1 encompassing the EXT1 and the adjacent TRPS1 gene. We report on a boy with an oligo array-cgh characterized small microdeletion involving EXT1 alone but with some features of Langer-Giedion syndrome suggesting a functional disturbance of TRPS1. This boy, in addition to a mild Langer-Giedion like phenotype, also had some unusual features including prominent toe pads and fat pads on the soles of his feet similar to those described in Pierpont syndrome.
Authors:
Jacqueline McBrien; John Anthony Crolla; Shuwen Huang; Jerry Kelleher; John Gleeson; Sally Ann Lynch
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Publication Detail:
Type:  Case Reports; Journal Article    
Journal Detail:
Title:  American journal of medical genetics. Part A     Volume:  146A     ISSN:  1552-4833     ISO Abbreviation:  Am. J. Med. Genet. A     Publication Date:  2008 Jun 
Date Detail:
Created Date:  2008-05-27     Completed Date:  2008-06-16     Revised Date:  -    
Medline Journal Info:
Nlm Unique ID:  101235741     Medline TA:  Am J Med Genet A     Country:  United States    
Other Details:
Languages:  eng     Pagination:  1587-92     Citation Subset:  IM    
Copyright Information:
2008 Wiley-Liss, Inc.
Affiliation:
Our Lady's Children's Hospital, National Centre for Medical Genetics, Dublin, Ireland.
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MeSH Terms
Descriptor/Qualifier:
Adipose Tissue / abnormalities
Child, Preschool
Chromosome Deletion*
Chromosomes, Human, Pair 8 / genetics*
DNA-Binding Proteins / genetics*
Humans
In Situ Hybridization, Fluorescence
Karyotyping
Langer-Giedion Syndrome / diagnosis*,  genetics*,  radiography
Male
N-Acetylglucosaminyltransferases / genetics*
Oligonucleotide Array Sequence Analysis
Phenotype
Toes / abnormalities
Transcription Factors / genetics*
Chemical
Reg. No./Substance:
0/DNA-Binding Proteins; 0/TRPS1 protein, human; 0/Transcription Factors; EC 2.4.1.-/N-Acetylglucosaminyltransferases; EC 2.4.1.224/exostosin-1

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


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