Document Detail


Severe clinical course of Hirschsprung disease in a Mowat-Wilson syndrome patient.
MedLine Citation:
PMID:  20145308     Owner:  NLM     Status:  MEDLINE    
Abstract/OtherAbstract:
We present a clinical case of a female infant with multiple anomalies and distinctive facial features, with an exceptionally severe clinical course of Hirschsprung disease. The girl was also diagnosed with Mowat-Wilson syndrome, confirmed by molecular analysis as a heterozygous deletion of the ZEB2 gene. Moreover, molecular karyotyping revealed a deletion involving further genes (KYNU, ARHGAP15, and GTDC1).
Authors:
R Smigiel; A Szafranska; M Czyzewska; A Rauch; Ch Zweier; D Patkowski
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Publication Detail:
Type:  Journal Article    
Journal Detail:
Title:  Journal of applied genetics     Volume:  51     ISSN:  1234-1983     ISO Abbreviation:  J. Appl. Genet.     Publication Date:  2010  
Date Detail:
Created Date:  2010-02-10     Completed Date:  2010-05-18     Revised Date:  -    
Medline Journal Info:
Nlm Unique ID:  9514582     Medline TA:  J Appl Genet     Country:  Poland    
Other Details:
Languages:  eng     Pagination:  111-3     Citation Subset:  IM    
Affiliation:
Department of Genetics, Wroc?aw Medical University, Marcinkowskiego 1, 50-368 Wroc?aw, Poland. smigiel@gen.am.wroc.pl
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MeSH Terms
Descriptor/Qualifier:
Abnormalities, Multiple / genetics*,  pathology
Female
GTPase-Activating Proteins / genetics
Gene Deletion*
Genotype
Glycosyltransferases / genetics
Heterozygote
Hirschsprung Disease / complications,  genetics*,  pathology
Homeodomain Proteins / genetics*
Humans
Hydrolases / genetics
Infant, Newborn
Karyotyping
Mental Retardation / complications,  genetics*,  pathology
Phenotype
Repressor Proteins / genetics*
Syndrome
Chemical
Reg. No./Substance:
0/GTPase-Activating Proteins; 0/Homeodomain Proteins; 0/Repressor Proteins; 0/ZEB2 protein, human; 0/rho GTPase-activating protein 15, human; EC 2.4.-/GTDC1 protein, human; EC 2.4.-/Glycosyltransferases; EC 3.-/Hydrolases; EC 3.7.1.3/kynureninase

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


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