Document Detail


Autosomal dominant syndrome of mental retardation, hypotelorism, and cleft palate resembling Schilbach-Rott syndrome.
MedLine Citation:
PMID:  19921646     Owner:  NLM     Status:  MEDLINE    
Abstract/OtherAbstract:
We present a family segregating for an autosomal dominant syndrome of hypotelorism, cleft palate/uvula, high-arched palate and mild mental retardation. Although these findings may suggest a form of holoprosencephaly, no holoprosencephaly was found on MRI of the proposita. Results of genetic studies were normal including FISH for deletion of 22q11, karyotype analysis, fragile X testing, high-resolution comparative genomic hybridization and SEPT9, SHH mutation analysis. The syndrome is reminiscent of the infrequently recognized autosomal dominant Schilbach-Rott syndrome.
Authors:
Vered Shkalim; Hagit N Baris; Gavriel Gal; Ruth Gleiss; Shlomo Calderon; Marja Wessels; Anneke Maat-Kievit; Bj??rn Menten; Elfride De Baere; Raoul C M Hennekam; Anja Schirmacher; Sherri Bale; Mordechai Shohat; Patrick J Willems
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Publication Detail:
Type:  Case Reports; Journal Article    
Journal Detail:
Title:  American journal of medical genetics. Part A     Volume:  149A     ISSN:  1552-4833     ISO Abbreviation:  Am. J. Med. Genet. A     Publication Date:  2009 Dec 
Date Detail:
Created Date:  2009-11-30     Completed Date:  2010-01-27     Revised Date:  -    
Medline Journal Info:
Nlm Unique ID:  101235741     Medline TA:  Am J Med Genet A     Country:  United States    
Other Details:
Languages:  eng     Pagination:  2700-5     Citation Subset:  IM    
Affiliation:
The Raphael Recanati Genetics Institute, Rabin Medical Center, Beilinson Hospital and Schneider Children's Medical Center of Israel, Petah Tikva, Israel.
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MeSH Terms
Descriptor/Qualifier:
Abnormalities, Multiple / genetics*
Adolescent
Cleft Palate / complications*,  genetics*
Comparative Genomic Hybridization
Cytoskeletal Proteins / genetics
Female
GTP-Binding Proteins / genetics
Genes, Dominant / genetics*
Hedgehog Proteins / genetics
Humans
Hypertelorism / complications*,  genetics*
Infant, Newborn
Male
Mental Retardation / complications*,  genetics
Middle Aged
Nuclear Proteins / genetics
Pregnancy
Syndrome
Twist Transcription Factor / genetics
Young Adult
Chemical
Reg. No./Substance:
0/Cytoskeletal Proteins; 0/Hedgehog Proteins; 0/Nuclear Proteins; 0/SHH protein, human; 0/TWIST1 protein, human; 0/Twist Transcription Factor; EC 3.6.1.-/GTP-Binding Proteins; EC 3.6.1.-/SEPT9 protein, human

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


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